PRECONCEPTION CARE

FROM POLICY TO PRACTICE AND BACK

SABINE FRANCISCA VAN VOORST

Preconception care: from policy to practice and back
PhD thesis, Erasmus University Rotterdam, The Netherlands

The research presented in this dissertation was performed at the department of Obstetrics and
Gynaecology, division of Obstetrics and Prenatal Medicine, Erasmus University Medical Centre,
Rotterdam, The Netherlands.

Part of the research in this dissertation was supported by a grant from the Ministry of Health,
Welfare and Sport (VWS), the Netherlands (grant 318804) and the National Organization for
Health Research and Development.

Financial support for the printing of this dissertation was kindly supported by:
Department of Obstetrics and Gynaecology, Erasmus MC, Nederlandse vereniging voor Obstetrie
and Gynaecologie (NVOG).

Copyright © 2017, Sabine Francisca van Voorst, the Netherlands

s.vanvoorst@erasmusmc.nl
Published manuscripts have been reproduced with explicit permission from the publishers. No part
of this thesis may be reproduced or transmitted in any form of written permission of the author.

Design: Wouter van Dijk

Printing: Optima Grafische Communicatie
ISBN / EAN: 978-94-92683-57-1
 PRECONCEPTION CARE
FROM POLICY TO PRACTICE AND BACK

Preconceptiezorg
Van beleid naar de praktijk en terug

Proefschrift
ter verkrijging van de graad van doctor aan de
Erasmus Universiteit Rotterdam
op gezag van de
rector magnificus
Prof.dr. H.A.P. Pols
en volgens besluit van het College voor Promoties.

De openbare verdediging zal plaatsvinden op

woensdag 14 juni 2017 om 11:30
door

SABINE FRANCISCA VAN VOORST

geboren te Rotterdam
PROMOTIECOMMISSIE

Promotoren: Prof.dr. E.A.P. Steegers

Prof.dr. S. Denktaş

Overige leden: Prof.dr. P.J.E. Bindels

Prof.dr. K. van der Velden
Prof.dr. R.M.W. Hofstra

Copromotor: Dr. L.C. de Jong – Potjer

Paranimfen: Dr. R. van Baars

Drs. V.L. van Voorst
TABLE OF CONTENTS
Chapter 1 General introduction 7

PART I Agenda setting and intervention selection

Chapter 2 Analysis of policy towards improvement of perinatal mortality in the 17
Netherlands (2004–2011)
Chapter 3 The Dutch national summit on preconception care: a summary of 41
definitions, evidence and recommendations
Chapter 4 Evidence-based preconceptional lifestyle interventions 59

PART II Designing and intervention approach

Chapter 5 The Healthy Pregnancy 4 All study: design and cohort profile 105
Chapter 6 Effectiveness of general preconception care accompanied by a 123
recruitment approach: protocol of a community-based cohort study
(the Healthy Pregnancy 4 All study)

PART III Implementation and evaluation

Chapter 7 Current practices of preconception care by primary caregivers in the 145
Netherlands
Chapter 8 Developing social marketed individual preconception care 161
consultations: which consumer preferences should it meet?
Chapter 9 Implementation of community-based peer health education strategy 179
for preconception care

PART IV Back to policy and practice

Chapter 10 General discussion 201
Chapter 11 Summary / Samenvatting 217

ADDENDUM
List of abbreviations 230
Authors and affiliations 231
List of publications 232
PhD Portfolio 236
About the author 241
Dankwoord 242
GENERAL INTRODUCTION
GENERAL INTRODUCTION

Preconception Care
Preconception care (PCC) is care for women or couples that contemplate pregnancy. It aims
to promote health of the future child by reducing or eliminating risks for adverse pregnancy
outcomes, prior to conception and in early pregnancy. Where risks cannot be reduced, PCC aims to
inform prospective parents to enable them to make informed decisions about pregnancy.
The content of PCC encompasses a vast amount of risk factors associated with adverse
perinatal health outcomes, which are important to address prior to conception to ascertain the
most benefit. These risk factors can be categorized within 13 domains: health care promotion
(e.g. unplanned pregnancy), immunization (e.g. inadequate protection against rubella), infection
(e.g. sexually transmitted diseases or toxoplasmosis), chronical medical conditions (e.g. diabetes),
psychiatric conditions (e.g. depression and anxiety disorders), maternal exposures (e.g. alcohol and
tobacco), genetic risks (e.g. genetic carriership of hemoglobinopathies), nutrition (e.g. obesity),
environmental exposures (e.g. solvents in paint), psychosocial stressors (e.g. domestic violence),
reproductive history (e.g. obstetric history of premature birth (<37 weeks of gestation)) and risks
within special groups (e.g. immigrant and refugee populations).1

The rationale for preconception care

Embryonic health is the basis for a healthy start in life, a healthy childhood and health in
adulthood.2-4 Preconception care is an essential addition to conventional perinatal health care for
several reasons.
Firstly, conventional antenatal care does not provide the opportunity for primary prevention.
It can only address risk factors when the foetus has already been exposed to risks for adverse
pregnancy outcomes as the first consultation during antenatal care occurs at best between the
8th and 12th week of pregnancy. By then, key events in embryonic growth and development
have already taken place. At about the 10th week of pregnancy approximately all organs and
the placenta have been developed. Developmental rates during the first trimester are even the
highest during ones’ entire lifetime.5 Not only are organs formed and does the embryo grow, foetal
programming occurs, during which functions of cells are determined. These three events are crucial
to the health of the foetus during pregnancy and its extra-uterine life. Addressing preconception
risk factors during antenatal care is simply too late, as preconception risk factors may already have
irreversibly affected embryonic health. These early exposures may give rise to the so-called ‘Big 3’
perinatal morbidities (small for gestational age (SGA), prematurity and congenital abnormalities),
which precedes mortality in 82% of the cases.6 It has been estimated that perinatal morbidity and/
or mortality can be reduced substantially with preconception care.7-10
Secondly, PCC can promote health in later life. Perinatal mortality and morbidity are the
first consequences of risk exposure in embryonic period. If preconception risk factors result in
permanent alterations in the structure and function of organs, the result of the affected embryonic
health is not only limited to perinatal mortality and morbidity, affected embryonic health can

8
contribute to higher risks for diseases in childhood and adulthood (e.g. risks for cardiovascular and
metabolic disease).11 PCC’s potential to prevent early exposure to risks provides an opportunity for
primary prevention of morbidity in later life.
Lastly, PCC can provide additional benefits for parental health. Becoming a parent can be
seen as an extra screening moment for health risks and can be an ultimate motivator to change
health behaviors. A well-known example is that many women say they will stop smoking if they are
pregnant. Smoking cessation reduces a woman’s’ risk of developing restrictive lung disease, (lung)
cancer and cardio metabolic diseases. In other words, utilizing the life event of parenthood can 1
provide a momentum for health promotion.

introduction
Organization of PCC in the Netherlands
It has been acknowledged to be a true challenge to select the optimal delivery strategy for PCC.12
In the Netherlands, the Health Council of the Netherlands has categorized preconception care into
collective preconception care (e.g. national campaigns) and individual preconception care (e.g.
consultations (risk assessment and consequent intervention during an individual consultation).13
Individual preconception care is subcategorized into general PCC (individual consultations for
women or couples with undefined risks) and specialized PCC (individual consultations for women
or couple with defined risks for adverse pregnancy outcomes).
Individual consultations provide the opportunity for professional led broad risk assessment.
Other forms often opportunistically address single risk factors when women present themselves
with specific questions or seek specific information. These forms therefore rely on women’s own risk
perception, which is known to be low.14 Given the advantages individual consultations, research
in this thesis focusses on organization and implementation of preconception care in the form of
individual consultations.

Point of departure for this thesis

The debate about the high perinatal mortality rate in the Netherlands as of 2004 ultimately
resulted in the awareness of the need to innovate in the organization of perinatal health care
and to emphasize preventive measures. Within this process, it was suggested that the nationwide
introduction of individual PCC consultation for the general public should be implemented in
primary care as of 2007.13
Although prerequisites were met for the delivery of general preconception care within primary
care (e.g. guidelines and risk assessment tools), nationwide introduction of individual PCC for the
general public was stalled. It was thought that more evidence was needed regarding whether PCC
approaches would reach high-risk women and would be effective in terms of risk reduction or
reduction of perinatal mortality.
In 2009 the Erasmus Medical Center initiated the Ready for a Baby Program or the ‘Klaar voor
een Kind’ program as it is referred to in Dutch. In this program new collaborations were formed
between the public health domain and caregivers from the curative health domain, to improve
perinatal health in disadvantaged neighbourhoods. Interventions were designed to address

9
each step in the chain of perinatal health: from the preconception phase to early childhood. The
experiences from this Rotterdam-based program provided the incentive to experiment with
programme-based preconception care, new risk selection during pregnancy and accessing high-
risk groups in other communities in the Netherlands nationally.15 This incentive met the agenda of
the Ministry of Health. In 2011 the Dutch Ministry of Health financed a national program to improve
perinatal health in disadvantaged neighbourhoods. This resulted in the Healthy Pregnancy 4 All
(HP4All) study. Given the insights in the importance of embryonic health and involvement of
public health, this program was designed to improve perinatal health by intervening before and
in early pregnancy. The program consisted of two interventions (1) programmatic preconception
care within primary care and (2) broadened risk selection with the Rotterdam Reproductive Risk
Reduction (R4U) instrument and multidisciplinary care pathways. Both interventions are evaluated
iteratively.
This thesis is based on studies conducted within or parallel to the HP4All – Preconception care
sub-study between (september 2011 – december 2014). This study formed the basis to reflect upon
the organization and implementation of preconception care in the form of individual consultations,
in this thesis.

AIMS OF THIS THESIS

The aims can be summarized as follows (see Figure 1):
• To evaluate the policy process and to review the evidence which led to selection of PCC as an
intervention to reduce perinatal mortality (Part I - Agenda setting and intervention selection).
• To develop a programmatic PCC intervention strategy in high risk municipalities – Healthy
Pregnancy 4 All (Part II – Designing an intervention strategy).
• To evaluate current practice and implementation of these strategies with involved stakeholders
(Part III –Implementation and evaluation).

THESIS OUTLINE
Part I – Agenda setting and intervention selection
Chapter 2 addresses the question how the agenda setting emerged and led to the policy of the
national government to intervene in the organization of perinatal healthcare to reduce perinatal
mortality and morbidity. In order to answer this question a retrospective policy analysis was
performed to investigate the process between 2004 and 2011.
Chapter 3 provides a summary of a National Summit on PCC in the Netherlands which took place
in 2012. During this summit professionals were engaged to reflect upon the available evidence and
knowledge gaps to frame future implementation of PCC.
Chapter 4 provides a thorough review of the evidence of lifestyle interventions. As lifestyle
interventions are applicable to a large proportion of couples in the general population, their
evidence is important to consider prior to the design of PCC programs.

10
 FIGURE 1: Contents and central philosophy of this thesis.
Perinatal mortality is only the tip of the iceberg. Being born with perinatal morbidity can give rise to disease and illness in
childhood and adulthood. Perinatal care should encompass preconception care (PCC) in order to promote perinatal health and
a healthy society. Implementation of individual PCC consultations was advocated in response to the disadvantaged position
of the Netherlands in perinatal health. With this thesis we look back upon the past decade and we draw lessons from research
regarding the organization of individual PCC within the Dutch primary care and public health system. Content of this thesis is
presented according to chronologic steps of a policy process depicted in this figure. Hence, the title of this thesis: ‘Preconcep-
tion care – from policy to practice and back’
1
I
AGENDA SETTING &
INTERVENTION SELECTION

introduction
BACK TO POLICY
& PRACTICE IV II DESIGNING AN
INTERVENTION STRATEGY

III IMPLEMENTATION
& EVALUATION

Part II - Designing an intervention strategy

Chapter 5 and 6 reflect upon how an intervention approach was developed to address risks before
and respectively during early pregnancy.

Part III – Implementation and evaluation

Within Part III the chapters reflect on what can be learned from the different stakeholders in
general preconception care: primary caregivers (midwives and general practitioners), women
contemplating pregnancy, municipal public health partners and peer health educators.
Chapter 7 focuses on what can be learned from the current preconception activities of primary
caregivers. A cross-sectional inventory is performed regarding their activities, perceptions and
prerequisites regarding delivery of (systematic) PCC in the future.
Chapter 8 looks into the preferences of the target population. A qualitative approach is used to
assess preferences across the four essential components of the social marketing approach (Product,
Place, Price and Promotion). Findings are important to tailor delivery of PCC consultations to the
needs and preferences of women.
In Chapter 9 an in depth study is presented regarding the implementation of peer health education
in preconception care. Recommendations are made regarding future implementation strategies.

11
PART IV - Back to policy and practice
After addressing each question in the main section of this thesis, the general discussion (Chapter
10) elaborates on principle findings and strengths and weaknesses of the methodology regarding
the organization of programmatic PCC in the future.

REFERENCES
1. Jack BW, Atrash H, Coonrod DV, Moos MK, O’Donnell J, Johnson K. The clinical content of preconception care: an overview
and preparation of this supplement. American journal of obstetrics and gynecology. Dec 2008;199(6 Suppl 2):S266-279.
2. Steegers EA. [Embryonic health and preconception care: importance for current and future generations]. Ned Tijdschr Ge-
neeskd. 2014;158:A7373.
3. van der Zee B, de Beaufort I, Temel S, de Wert G, Denktas S, Steegers E. Preconception care: an essential preventive strategy
to improve children’s and women’s health. J Public Health Policy. Aug 2011;32(3):367-379.
4. Templeton A. The public health importance of antenatal care. Facts, views & vision in ObGyn. 2015;7(1):5-6.
5. Schoenwolf GC, Larsen WJHe. Larsen’s human embryology. 4th ed., Thoroughly rev. and updated. ed. Edinburgh: Churchill
Livingstone; 2009.
6. Bonsel GJ, Birnie E, Denktas¸ S, Poeran J, EAP S. Dutch report: Lijnen in de Perinatale Sterfte, Signalementstudie Zwanger-
schap en Geboorte 2010 . Rotterdam: Erasmus MC; 2010. Available at: www.nvk.nl/Nieuws/Dossiers/Dossier Perinatalezorg.
aspx. Retrieved January 5, 2011.
7. Wildschut HI, van Vliet-Lachotzki EH, Boon BM, Lie Fong S, Landkroon AP, Steegers EA. [Preconception care: an essential
part of the care for mother and child]. Ned Tijdschr Geneeskd. Jun 17 2006;150(24):1326-1330.
8. Shannon GD, Alberg C, Nacul L, Pashayan N. Preconception health care and congenital disorders: mathematical modelling
of the impact of a preconception care programme on congenital disorders. BJOG : an international journal of obstetrics
and gynaecology. Apr 2013;120(5):555-566.
9. Czeizel AE. Ten years of experience in periconceptional care. European journal of obstetrics, gynecology, and reproductive
biology. May 1999;84(1):43-49.
10. Macklon NS, Geer, I.A., Steegers, E.A.P. Textbook of Periconceptional medicine. . London: Informa Healthcare; 2009.
11. Barker DJ, Thornburg KL. The obstetric origins of health for a lifetime. Clinical obstetrics and gynecology. Sep 2013;56(3):511-
519.
12. Shannon GD, Alberg C, Nacul L, Pashayan N. Preconception healthcare delivery at a population level: construction of public
health models of preconception care. Matern Child Health J. Aug 2014;18(6):1512-1531.
13. Health Council of the Netherlands: Preconception care: a good beginning Health Council of the Netherlands. The Hague
2007. publication no. 2007/19E, ISBN 978-90-5549-657-0.
14. Hosli EJ, Elsinga J, Buitendijk SE, Assendelft WJ, van der Pal-de Bruin KM. Women’s motives for not participating in precon-
ception counseling: qualitative study. Community genetics. 2008;11(3):166-170.
15. Denktas S, Bonsel GJ, Steegers EA. [Perinatal health in Rotterdam, the Netherlands--experiences after 2 years of ‘Ready for a
baby’]. Ned Tijdschr Geneeskd. 2012;156(29):A4289.

12
1

introduction

13
 PART I

1
AGENDA SETTING &
INTERVENTION SELECTION
ANALYSIS OF POLICY TOWARDS
IMPROVEMENT OF PERINATAL
MORTALITY IN THE NETHERLANDS
(2004 - 2011)
S.F. van Voorst‡, A.A. Vos‡, E.A.P. Steegers, S. Denktaş
‡ Shared first authorship
Social Science & Medicine 2016; 57: 156-164
ABSTRACT
Objectives: Relatively high perinatal mortality and morbidity rates in the Netherlands resulted in
a process which induced policy changes regarding the Dutch perinatal healthcare system. Aims of
this policy analysis are (1) to identify actors, context and process factors that promoted or impeded
agenda setting and formulation of policy regarding perinatal health care reform and (2) to present
an overview of the renewed perinatal health policy.
Methods: The policy triangle framework for policy analysis by Walt and Gilson was applied.11
Contents of policy, actors, context factors and process factors were identified by triangulation of
data from three sources: a document analysis, stakeholder analysis and semi-structured interviews
with key stakeholders.
Results: Analysis enabled us to chronologically reconstruct the policy process in response to the
perinatal mortality rates. The quantification of the perinatal mortality problem, the openness
of the debate and the nature of the topic were important process factors. Main theme of policy
was that change was required in the entire spectrum of perinatal healthcare. This ranged from
care in the preconception phase through to the puerperium. Furthermore emphasis was placed
on the importance of preventive measures and socio-environmental determinants of health. This
required involvement of the preventive setting, including municipalities. The Dutch tiered perinatal
healthcare system and divergent views amongst curative perinatal health care providers were
important context factors.
Conclusions: This study provides lessons which are applicable to health care professionals and
policy makers in perinatal care or other multidisciplinary fields.

18
INTRODUCTION

The health issue

Several studies have revealed that the Netherlands has relatively unfavorable perinatal mortality
rates.1-3 In 2004, the PERISTAT I study showed that the Netherlands was one of the European
countries with the highest perinatal mortality rates (10.5 mortality cases per 1000 births as of 22
weeks of gestation).3 The Dutch position slightly improved in 2008 (10 mortality cases per 1000
births), but rates remained relatively high compared to other European countries.3 In response,
numerous studies were conducted to identify causes and determinants of perinatal mortality. It
became clear that there were large perinatal health inequalities within the country, which were 2
associated with low socioeconomic status. 4-8

A policy analysis of perinatal health care

Whilst it is widely acknowledged that poor socioeconomic circumstances affect health
throughout life, it was only during the last decade that this concept was translated into actual policy
regarding perinatal healthcare in the Netherlands. Due to these numbers, the unique organization
of perinatal care in the Netherlands was questioned in the open for the first time.
The concern of relatively high perinatal mortality and morbidity rates triggered a policy process
which resulted in intervention in the organization of perinatal health care.

The health policy environment

The policy process took place within a uniquely organized field: the Dutch perinatal health system.
Figure 1 depicts the field that is involved in either formulating or implementing perinatal health
policies.

The perinatal healthcare field

The curative care echelon: Curative care in the perinatal healthcare field is comprised of
preconception care, antenatal care, labor care and postpartum care. This care is delivered by
primary caregivers (midwives in the community), secondary caregivers (gynecologists in secondary
hospitals) and tertiary caregivers (gynecologists in university hospitals). Inherent to the Dutch
system, gynecologists are also obstetricians. Professionals within these three tiers function
autonomously in accordance to their own guidelines. Co-operation between the professionals of
the tiers is close because of delineation by the so-called ‘List of obstetric indications’, or LOI. This
consensus based list provides indications to allocate women to care according to either a ‘low risk’
for pathology or a ‘high risk’ for pathology. When allocated to the low-risk category, women receive
care exclusively from a community midwife and can opt for a home birth or out-patient hospital
birth. High risk women are attended to by gynecologists or clinical midwives in the second or third
tier, they do not have the option of a home-delivery.
The organization model of perinatal care of the Netherlands differs from other countries
because of the strong and independent position of midwifery.9,10 Firstly midwives have power
because in the Dutch system they attend to the largest proportion of pregnant women (80% of
pregnancies start in the first tier of care). The fact that women typically start antenatal care with a

19
 FUNDING
Of curative care
National Health-insurance system
POLITICAL SYSTEM
Municipal level

● Municipal
CURATIVE CARE ECHELON ● Department of Public Health
government
Preconception care – Antenatal care – Labor care – Postpartum care PUBLIC HEALTH CARE ECHELON
● Primary care – Midwifes Nationally advocated Municipal advocated
National level
● Prenatal screening Program for ● Education on
● Secondary care – Obstetricians Maternity care Infectious diseases & pregnancy and childbirth ● Parliament
Erythrocyte immunization ● Local policy
● Tertiary care – Obstetricians ● Prenatal Screening for
trisomies ● Department of curative
● 20 week anomaly ultrasound ● Ministery health
Municipal health service
of Health

20
● Department of public
health

● Other ● Other departments/

ministries staff directories
FIGURE 1: The health policy environment (adapted from Schafer et al. 2010

SUPPORT, RESEARCH & DEVELOPMENT, MONITORING

26
)

Academic setting Profesional organisations Monitoring institutes Advisory institutes

● Departments of Obstetrics / Gynaecologie
of University hospitals
● Midwifery research network
● Dutch Society of obstetrics and
Gynaecology; Royal Dutch Medical
Association; Association of Medical
specialists
● Royal Dutch Association of Midwives
● Dutch Healthcare Authority
● Health care Inspectorate
● Organization for Health Research and Development
● The Netherlands Organization for Applied Scientific
Research
● Dutch Institute for research in health care
● National Insitute for Public Health and the Environment
● Statistics Netherlands
● Health Council Netherlands
midwife is driven by the Dutch culture in which childbirth is seen as a natural process. Furthermore,
the ‘LOI’ defines indicated care in secondary and tertiary settings. The latter is only reimbursed after
referral by primary care.
Secondly, the government provides favorable licensing laws, insurance regulations and
government support for midwifery education.9 Lastly, the professional organization of midwives
safeguards autonomous roles of midwives.9,10 The autonomy and the large role of midwives is seen
as one of the reasons that the Netherlands is a country with relatively non-medicalized birth, with
low rates of obstetric interventions (e.g. caesarean section) and high rates of home-births.
The public healthcare echelon: General public healthcare – also referred to as the preventive
health care setting - is organized at a municipal level except for a few elements of preventive 2
perinatal care, which are organized nationally (the screening of infectious diseases and erythrocyte

A policy analysis of perinatal health care

immunization, first trimester screening for chromosomal abnormalities and ultrasound detection
of fetal anomalies). At municipal level, the public health departments are responsible for the
organization of disease prevention, health promotion, and health protection. Involvement of the
municipal public healthcare setting in perinatal healthcare was often restricted to promotion of
lifestyle in pregnancy and information about breastfeeding.

Support, research and development

The curative perinatal healthcare field has an extensive infrastructure for research and development.
Besides the scientific expertise from universities, there are several private and government led
institutes that independently advise the sector. Professionals in the curative perinatal healthcare
field are represented by professional organizations. Midwives have their own professional
organization, The Royal Dutch Association of Midwives, which aims to strengthen the independent
position of midwives by promoting the quality and access to midwifery-led care. Gynecologists
are represented in three professional organizations, namely the Royal Dutch Medical Association,
Association of Medical Specialists, and the Dutch Society of Obstetrics and Gynecology. The latter is
responsible for developing guidelines.

Funding of the system

The curative health system is funded by national health-insurance. Basic health insurance covers
all essential curative care. Its content is regulated by the Health Insurance Act. Additional to basic
health insurance, insurance companies provide supplementary packages.

Political system and development of health policy

In the Netherlands, health policy is made at national, provincial and municipal levels.
Since 2006 the national government’s role in health care policy has changed. Instead of being
responsible for direct control of volumes, prices, and productive capacity, the national government
fulfills a regulatory and supervisory role. Most of the tasks are delegated to independent bodies.
Public health care policy is defined by the Health Ministry, Welfare and Sports (from now on called
the Health Ministry) with a national memorandum on public health. This memorandum is written

21
every 4 years by the department of public health – a sub-department of the Health Ministry.
Different institutes provide local statistics to identify health and environmental issues which need
to be addressed with policy in the memorandum. To shape national policy to meet local needs of
municipalities, each municipality writes an additional memorandum. This enables municipalities
to deviate from the national memorandum whenever local environmental statistics point out
additional needs.

Changes in perinatal healthcare policy

Before 2004, effectuation of perinatal health policy largely depended on the curative system and
its own research and development field. Collaboration between policy makers of the departments
of curative and public health of the Health Ministry was uncommon. Retrospectively, the perinatal
mortality debate has led to policy reform, to many initiatives and to mind switches (e.g. to address
socio-economic determinants) in the field. These changes are in stark contrast to the culture prior
to the debate, when the way perinatal health care was organized was undisputed.
By benchmarking perinatal mortality and morbidity rates, EURO-PERISTAT has exposed that
several European countries have relatively unfavourable perinatal health statistics. Ideally, this
benchmarking would result in policy changes to improve perinatal health in these countries.
The EURO-PERISTAT group has even formulated the goal to monitor policy initiatives over time.3
Comparison of policies in response to high perinatal mortality and morbidity rates is informative
for countries facing the challenge to improve perinatal health at a population level. This paper
provides a retrospective analysis on the policy process that was brought about by publication of
perinatal mortality rates by EURO-PERISTAT. To our knowledge this is the first policy analysis in
the literature aiming to summarize the policy measures that have been taken after the perinatal
mortality debate took flight after the EURO-PERISTAT reports.
With our policy analysis we return to the beginning of the perinatal mortality debate in 2004
and we focus on the policy process and the proposed measures to reduce perinatal mortality and
inequality. The aim of this policy analysis was twofold. First, we evaluate ‘why’ and ‘how’ the perinatal
mortality problem made its way onto the political agenda. Secondly, we present an overview of
the overall contents of the renewed perinatal health policy. In the discussion, we elaborate about
lessons which can be drawn from this policy process.

METHODS
In this evaluation we applied the Policy Triangle framework for policy analysis by Walt and Gilson
(see Supplementary Figure 1).11,12 The variables of this policy triangle (actors, content, context and
process) formed the basis for our data collection and organization. Data collection was conducted
retrospectively (initials removed). The timeframe of the analysis is from 2004 to 2011.
• Document analysis: An electronic search was performed in the database of the Dutch
government to identify documents about perinatal mortality.13 The search was performed
for the period of January 2004 to January 2012. Keywords were: Pregnancy, Perinatal Health

22
 and Preconception care. Documents that reported high perinatal mortality rates or about
interventions to reduce perinatal mortality were eligible for the document analysis. Two
authors (initials removed) assessed eligibility of identified documents. They performed citation
tracking; meaning they collected the grey literature (e.g. newspaper articles or scientific
publications) cited by important documents. The document analysis provided potential actors
and key content of the policy triangle.
• Stakeholder analysis: We defined stakeholders as those individuals or organizations with an
interest in an issue or policy, those who might be affected by a policy and those who may play
a role in making the policy.14 A list of stakeholders, their positions and interests with respect to
perinatal mortality was made, based on recollection of the authoring team and the document 2
analysis. Key informants were selected by consensus of the authoring team.

A policy analysis of perinatal health care

• Interviews: The key informants identified in the stakeholder analysis were interviewed in order
to investigate the policy process. Additional interview candidates were identified during the
process. The candidates varied from scientists in the curative sector to representatives of the
professional organizations to delegates of the national Health Ministry and the Dutch House of
Representatives. Interviews were performed according to a semi-structured topic list consisting
fixed format and open questions. The interview item list consisted of the following domains: (1)
inquiry regarding position and interests to verify the stakeholder analysis, (2) agenda setting
and (3) intervention selection. Across these domains questions were formulated to identify
elements of the policy triangle (actors, content, context and process factors). The interviews
were transcribed verbatim. Fragments of the interviews were coded into elements of the
policy triangle (actors, context, content or process) by two authors (initials removed). Interview
candidates provided consent for the use of citations.
Elements of the policy triangle (fragments or summaries of interviews or documents) were extracted
and classified as ‘actor’ (individuals or organizations that affect policy), ‘content’ (substance of
a particular policy which details its constituent parts), ‘context’ (political, economic, social or
cultural factors which may have an effect on health policy) or ‘process’ (the way in which policies
are initiated, developed or formulated, negotiated, communicated, implemented and evaluated).
These items were organized chronologically in a data spreadsheet. This coding approach was
piloted after which the two data extractors (SVV and AV) had consensus on the approach. This
allowed us to make a chronological reconstruction of the policy process structured by the elements
of the policy process.

23
RESULTS
The search of our document analysis identified 437 relevant hits of which 64 hits were included in
the document analysis (379 hits were excluded after retrieval of the document in case of duplicates
or because the topic was not related to perinatal mortality).
The results of the stakeholder analysis are provided in Supplementary Table 1. All (delegate(s)) of
the identified organizations in were approached. All approached individuals agreed to participate
in the interviews. In total 12 interview candidates were interviewed in 9 sessions (individually or in
pairs) varying from 30 – 90 minutes.
The identified content of governmental policy was organized in a chronologic time line
(Figure 2). This formed the basis for the chronologic headings according to which we described
the policy process in this section. Actors, content, context and process factors of this chronologic
reconstruction were summarized graphically in the policy triangle in Figure 3.

FIGURE 2: Time line of governmental measures to improve perinatal outcomes in 2004-2011

2004 Peristat I report

2005 The government instructs to re-examine the position of

Measures to prevent perinatal mortality:
- Systematic monitoring of perinatal health outcomes
- Introduction of a perinatal audit
- Extensification of screening in pregnancy
- Protocolled maternity care
- Extended screening with the Guthrie test
- Studies to investigate trends in perinatal mortality
and underlying causes
the Netherlands’ perinatal mortality rates
Pilot study on a perinatal audit system to review
perinatal mortality cases
2006 One of the opposition parties proposes a set of measures
to reduce perinatal mortality rates

2007 Preparation of a new set of measures

Outlines renewed antenatal policy:

2008 Peristat II report
- Preconception care Announcement of policy plan to reduce differences in health
- Institution of Steering Group outcomes by addressing socio-economic disparities
- Antenatal health care Announcement of renewed antenatal policy
- Postnatal care 2009 Institution Advisory Committee Pregnancy and Birth
- Recognition that external factors
influence perinatal mortality Institution of the National Perinatal Audit (PAN)

Core teams report Steering Group: 2010 Steering Group advice (report)
- Need for organizational improvement
- Enhancement of quality of care
- Improvement of risk assessment
- More attention to health education
and lifestyle (especially in deprived areas) 2011 Institution College of Perinatal Health (CPZ)
Start national program: Healthy Pregnancy 4 All

24
Policy process

First political measures: 2004 – 2008

Up until 2008, perinatal health care was a low priority file to the department of curative care
(within the Health Ministry). The file was dormant as there were no large issues within the perinatal
health field until the perinatal mortality rates became apparent. According to the interviewed
candidates, policy makers were surprised with the perinatal mortality statistics. They perceived
that the Netherlands had one of the best perinatal health care systems of Europe. All interviewees
confirmed that the results of the first PERISTAT report received little attention from politicians, the
professional organizations and health care providers. Interview candidates explained that at first 2
the perinatal mortality statistics were not perceived as a problem because the credibility of the

A policy analysis of perinatal health care

data was debated. The general opinion of the field was that the unfavorable perinatal mortality
rates of the Netherlands in comparison to other countries were due to underreporting of perinatal
mortality and morbidity rates in other countries. Furthermore, explanations were sought in specific
characteristics of the Dutch population (e.g. the relatively older age of future mothers).15 In 2005
the Minister of Health asked the National Institute for Public Health and Environment to verify
the outcomes of the first PERISTAT report.13 They concluded that the increased perinatal mortality
was at least partly explained by factors that can be improved by more effective prevention (i.e.
preconception care, smoking cessation). The Health Ministry remained expectative. This led
to parliamentary questions. One member of the House of Representatives was particularly
dedicated to midwifery-led perinatal care. Perinatal health issues were often directly associated
to this member. In the absence of measures from the Health Ministry, the political party of this
representative proposed a set of measures to improve perinatal mortality rates in the Netherlands
in 2006.13 In response the Health Ministry introduced an initial set of measures to lower perinatal
mortality rates and to improve perinatal health (see Figure 2) for the contents of the measures.13
These measures were largely similar to the measures proposed by the political party of the
aforementioned representative. In the meantime this party had become a governing party rather
than an opposing party. This provided them with more power. The first preventive measures which
were introduced up to 2007 included: 1. systematic monitoring of perinatal health outcomes by a
national perinatal database; 2. introduction of a perinatal audit; 3. increased screening in pregnancy;
4. protocolled maternity care; 5. adding diseases to the neonatal screening program (Guthrie test)
and 6. commissioning the Netherlands organization for Health Research and Development to set
up a research program to investigate trends in perinatal mortality and underlying causes. This came
to be the Perinatal Audit. They started to audit term perinatal mortality cases as of January 2010.

25
 NATIONAL POLICY TRIANGLE

CONTEXT

ACTORS
National Government
· Health Ministry
· Department of Public Health and
Curative health
· Interdepartmental network: ‘baby club’
· Politicians in the Dutch
House of Representatives
· Advisory Committee on Pregnancy
and Childbirth
Perinatal healthcare field
· Gynaecologists, Midwives
· Professional organizations of
Gynaecologists and Midwives
· Academic field
· EURO PERSITAT Group
Media
CONTENT PROCESS

CONTENT PROCESS CONTEXT

First measures before 2008

• Expectant management towards Peristat I • Peristat I • Nature of the subject

report • Reconsideration of Dutch Perinatal mortality • Obstetrics was a low priority topic for the
• Suggestion of measures by a dedicated rates department of Curative Health.
member of House of Representatives Plan to • Resistance by the political opposition party to
found the first collaborative platform for the expectant management of the Health
obstetric care. Ministry
• Plan to found the Perinatal audit
• Plan to found the Dutch Perinatal Registry
(PRN)
• First policy measures (see Figure 2)

Renewed perinatal healthcare policy and translation to practice 2008 - 2011

• Formation of the Steering Group
• Allocation of financial resources
• Renewed antenatal policy: intervention in care
(curative and preventive health care)
throughout the whole range of perinatal health
care.
• Formation of College of Perinatal Health to
monitor effectuation of measures proposed by
the Steering Group
• Commissioning of the ‘Pregnancy and
Childbirth’ research program
• Commissioning of the Healthy Pregnancy 4 All
program
• Pivot point: recognition of the perinatal
mortality problem after Peristat II
• Political interference: parliamentary questions
required rapid actions
• Incontrollable media attention
• Founding of the 'Babyclub'
• Mind shift from curative to preventive care,
emphasis on municipal involvement, as the
directory of preventive care picks up the
perinatal mortality problem.
• Participation of different professionals in the
Steering Group to select interventions thought
the whole spectrum of perinatal care.
• Collaboration between the Department of
Public Health and the Erasmus MC to develop
an intervention approach
• Unique organization of perinatal health care in
the tiered Dutch Health care system
• Different key principles between
Gynecologists and Midwifes gave friction.
• Perinatal health was not embedded as a
responsibility of the public health care field.
• Trend in community based intervention
approaches and effects of physical and social
environment on health.
• Exemplary intervention program: Ready for a
Baby (see Municipal policy triangle).
• Repetitive demission of cabinet resulting in
shifts in political coalitions from left to liberal
wing parties.

26
 MUNICIPAL POLICY TRIANGLE

CONTEXT
The Municipality of
Rotterdam had an own
policy process with regards
to their high perinatal
mortality and morbidity rate
This process is an
important context factor for
the policy triangle at a
national level depicted ACTORS
2
on the previous page.

A policy analysis of perinatal health care

Muncipal Government
· Aldermen
Preventive health care field
· Municipal health service
Curative health care field
· Gynaecologists, Midwives
Perinatal healthcare field
· Gynaecologists, Midwifes
· Academic field Erasmus MC
Lobbyists
· Social Platform Rotterdam
Media

CONTENT PROCESS

CONTENT PROCESS CONTEXT

• Allocation of resources to effectuate the
proposed initiatives to reduce perinatal
mortality.
• Commissioning of the Ready for a Baby
program.
• Urban perinatal health inequalities and need • High urban perinatal mortality rate and
of organizational change in perinatal health concomitant problem: safety and capacity
care was recognized by the academic problems in the midwifery field
department of Obstetrics. • Perinatal health (care) became a concern for
• Bridging between research field and a social municipal policy makers
lobby platform towards municipal policy • Media attention for municipal problems and
makers brought the perinatal mortality solutions
problem on the agenda. • Fast translation from science to care due to the
• Creating engagement amongst curative and nature of the problem
preventive sector to collaborate in a municipal
based intervention program.
• Selection of interventions in different phases
of perinatal health care
• Transparent program out roll and the novelty
of the approach promoted that the program
was noticed by national policy.

FIGURE 3: P olicy process: The policy triangle was used to summarize the policy process
according to the policy triangle.

27
Renewed perinatal healthcare policy 2008 – 2010
The PERISTAT II report was a turning point in perinatal health care policy.1 The Netherlands was
confronted with its disadvantaged position in Perinatal Health compared to other European
countries for a second time. As a consequence, the Dutch PERISTAT project group and both
professional organizations consciously decided to inform the media to achieve political agenda
setting. The media eagerly joined the debate. All respondents provided that this was due to the
nature of the topic. One respondent stated: “The media invented the term ‘baby mortality’ instead
of ‘perinatal mortality’, since that is a term that the public is not familiar with. The problem with baby
mortality is that is has connotations of healthy babies dying, instead of premature or intra-uterine
death.” Furthermore, perinatal health is an accessible topic: “almost everybody has an opinion
about the topic: birth. Everybody knows somebody who has had a baby, even if they haven’t
had one themselves. Everybody can condemn a ‘baby death rate.” Although the media attention
appeared to be a useful tool in the beginning, it was felt that the attention became incontrollable
and the nuances in the perinatal mortality debate were lost: “the debate took on a life of its own,
and then of course you cannot get the genie back into the bottle.” The debate diverted to looking at
causes for perinatal mortality within the care system – as the way perinatal care is organized in the
Netherlands is the most obvious difference with other European countries.

An intense debate between the professional organizations and the step to agenda setting
Whilst professional organizations were paralyzed by disagreement when they were first confronted
with the perinatal mortality statistics in 2008, they were forced to agree to work together to reduce
perinatal mortality. Interviews confirmed that from this time on both professionals organizations
gave the perinatal mortality issue a high priority. However, collaboration between the two was
complicated because of the historical incongruous visions of how perinatal health care should be
organized optimally. Proposed solutions concentrated on the integration of antenatal care.13
The risk approach of gynecologists is not always accepted by midwives. Midwives believe
the proactive approach of gynecologists to risk in childbirth leads to unnecessary obstetric
interventions. This medicalization of pregnancy is in direct contradiction with the philosophy that
birth is a natural process and with client centeredness. According to midwives “A pregnant woman
is not a patient to a midwife, for the midwife the pregnant woman is a client: a woman that happens
to be pregnant rather than a pregnant patient that happens to be a woman.” Aspects regarding
risk assessment are a classic dispute between midwives and gynecologists in the Netherlands.
According to gynecologists, risk assessment was failing and the dichotomous categorization into
high and low risk was failing. “In practice the difference between high- and low-risk patient is
difficult to preempt: in the end somebody in the low-risk compartment can have complications and
it occurs that somebody in the high-risk category does not have complications.”
Altering the approach to risk assessment touches concepts of ‘professional autonomy of
midwives’ and medicalization (“an increase of ‘risk thinking’ legitimates more medicalization”) and
client centeredness.
Another factor that contributed to incongruous visions was the different perspectives on issues

28
of the professional organizations due to their different goals. The professional organization of
gynecologists is primarily tasked with providing evidence based guidelines while the safeguarding
of the professions interests is delegated to a different organization. In contrast, the professional
organization of midwives has both of these roles. They were originally founded to safeguard the
position of the midwifery profession. Later they also became responsible for the development of
guidelines. Interviews confirmed that this provided an unequal position in the discussion, as this
resulted in the questioning of their objectivity in the debate.
Another factor in the policy process was the high public profile of the debate. Both professional
organizations stated that attention form the media initially had a positive effect. It provided
urgency to address the debate and achieve a consensus regarding the need to intervene in 2
perinatal health. However, later in the debate, the same attention from the media was reported as

A policy analysis of perinatal health care

an impediment. The professional organization of midwives stated that the speed and negativity
of the media required a defensive stance, in which they lost time to gain a proactive mode to
formulate measures.
Contrary to what one would assume, it was not the professional organizations themselves or
the Health Ministry that raised the urgency of addressing the perinatal mortality issue. Rather it
was parliament that once again insisted upon a rapid response to resolve the perinatal mortality
problem. Due to 17 parliamentary questions perinatal mortality became a ‘key priority’ at the Health
Ministry. Parliamentary questions are questions asked by a member of parliament to the Minister in
relation to parliamentary law and political decisions. These questions are answered during debates
and by means of letters. Parliamentary questions are thus a way to get items on the political
agenda and to initiate actions by departments.13 Meanwhile, the aforementioned Representative
remained to represent the interest of community midwives. Due to the urgency of the problem a
so-called ‘Baby club’ was initiated at the Ministry of Health. This unique interdepartmental network
provided a platform to discuss solutions with policy makers from different departments of the
ministry. This enabled them to answer the parliamentary questions rapidly. The functioning of the
interdepartmental network was even referred to by one respondent as “disaster command center.”
The National Institute for Public Health and the Environment used the national perinatal
database to confirm the PERISTAT II data and to reveal potential underlying causes of adverse
outcomes. They identified causes within four categories: 1. organization of perinatal care (e.g.
travel time to a hospital, collaboration between community midwives and gynecologists or risk
assessment), 2. maternal factors (e.g. ethnicity or education level), 3. fetal factors (e.g. congenital
anomalies), and 4. socio-demographic factors (e.g. deprived area).13 Recognition of these potential
causes shed the light on two main themes. Firstly, interventions were necessary within the
entire perinatal health care system from the preconception period through to and including the
puerperium. Secondly, the role of non-medical risk factors and the influence of neighborhood
deprivation on perinatal mortality was recognized to be more important than previously
thought.16,17
The Minister of Health needed to come up with rapid measures due to the urgency of the
issue created by the House of Representatives by means of parliamentary questions. The Health

29
 Ministry presented five main intervention
BOX 1: Renewed perinatal policy
themes (based on prior inquiries) in 2008
1. Preconception care: stimulating folic acid intake; explore which are presented in Box 1 and planned to
the efficacy of general perception and eventually inte- install a Steering Group to refine the strategy
grate this in the health care system. to intervene in the organization of perinatal
2. Institution of Advisory Committee: advice on quality-en-
health care the Steering Group was installed
hancing measures for the entire obstetric chain (from pre-
in 2008.13 Specific aims of the Steering group
conception care to maternity care), with special attention
to deprivation, organization of care and development of
were: to investigate whether the PERISTAT
guidelines. results were correct, to identify potential
3. Antenatal health care: introducing quality indicators, in- causes for the higher perinatal mortality
vestigation hospital performance at off business hours, rates and to propose specific measures. The
special attention to care in deprived neighborhoods
Health Ministry placed particular value on
(safety and extra tariff ).
the advices forthcoming from this committee
4. Postnatal care: evaluation the current capability of ma-
ternity care, evaluation the implementation of extended
and postponed actions until this committee
neonatal screening. completed its investigation.13 Two years
5. External factors: reduction socio-economic related health after its installation, the Steering Group
differences presented a comprehensive report which
was widely accepted by the field.18 The key
recommendations of this report were largely
in line with the previous policy changes (see box 1), but translated into more practical measures:
the need for organizational improvement, improvement of quality of care (in particular care in
acute situations), improvement of risk assessment, and more attention to health education and
lifestyle in and before pregnancy with a focus on deprived areas.13
The Minister of Health adopted the plans. However, when the cabinet fell, effectuation of the
advices was delayed. This is when the department of public health and the Ministry of Living, Work
and Integration became involved to implement the advices of the Steering Group. From this point
on, the direction of curative care of the Health Ministry took a step back.

Simultaneous actions at a municipal level

A local policy process took place in the municipality Rotterdam (see municipal policy triangle in
Figure 3). This process was a contextual factor for the national policy process. Rotterdam had a
perinatal mortality rate far above the country’s average. The local department of Obstetrics and
Gynecology approached a local social platform (an advisory board for societal issues towards
politics) to discuss the municipality’s inequality in perinatal health and potential solutions with the
local Alderman. Once it was realized at municipal level that “you need a healthy start in life to have
a healthy society” the agenda was set. Policy makers took on this vision and wrote a memorandum
in which they stated the intention to reduce perinatal mortality and morbidity rates to the national
level within 10 years.19 The municipality provided budget to effectuate the proposal which resulted
in the ‘Ready for a Baby program’ as of 2009.20 The perinatal mortality problems and solutions in
the ‘Ready for a baby’ program caught attention from the Ministry of Health and the House of

30
Representatives. Later it proved to be exemplary in the effectuation of urban perinatal health policy.

From policy to practice 2010 – 2011

In this period, the department of public health (of the Ministry of Health) became engaged in the
perinatal mortality debate. Prior to 2010, perinatal mortality was predominantly seen as a topic for
the curative sector and not seen as an item for the public health sector. However, the department
of public health persuaded the Health Minister that improvement of perinatal mortality required
their involvement. They had the vision that health care should be delivered locally and that ‘health
should be seen in relation to the social and physical environment’. This was taken up in the national
policy memorandum. It was seen as a challenge to spread this message and to make perinatal 2
health a key priority amongst municipal health policy makers. Furthermore, to intervene in perinatal

A policy analysis of perinatal health care

health collaboration between curative and preventive domains within local municipalities was
needed. The department of public health identified the need for a dedicated project to effectuate
their vision. The department of public health persuaded the Minister of Health to allocate extra
budget to develop evidence based interventions to reduce perinatal morbidity and mortality
rates. In agreement with their vision that policy should address socio-environmental health
factors, the intervention would require implementation of preventive measures at municipal level.
They recognized that they needed a field partner to engage municipalities in effectuating their
vision and evaluating their vision with research. They identified the ‘Ready for a Baby’ program in
Rotterdam. Having approached its executors, namely the Department of obstetrics of the Erasmus
University Hospital, they discussed a national program. This resulted in a research proposal and
grant to facilitate what later became the Healthy Pregnancy 4 All program. Two interventions were
selected: 1) a preconception care program in which both curative and preventive professionals
participate and 2) systematic risk assessment with an antenatal risk assessment tool addressing
medical and non-medical risk factors and associating care pathways.21,22 The program was launched
in the deprived neighborhoods of 14 municipalities with the most adverse perinatal outcomes,
compared to the national average.5 The effectiveness of the implementation of two interventions
in the local municipal setting was to be assessed by research parallel to the program.5 In total, the
Health Ministry provided 9 million euro for the entire period to effectuate the proposed measures
to intervene in perinatal healthcare. A board, namely the College of Perinatal Health, was formed to
superintend the effectuation of the measures of the Advisory Committee.13

Perinatal mortality debate: a catalyst to innovate

Retrospectively, this policy process in response to perinatal mortality provided several side effects
that resulted in additional events and interventions in the perinatal healthcare field. A selection of
these additional events is presented in Supplementary Box 1.

31
DISCUSSION

Main findings
The Perinatal mortality was a health issue which resulted in a national policy process. In this study
we performed a retrospective analysis regarding the agenda setting and formulation of policy to
intervene in the perinatal mortality rate between 2004 and 2011.
Attention for the topic resulted in the creation of a new network of policymakers consisting of
policymakers from both the department of curative and preventive health, politicians, researchers
and practitioners. This resulted in the review of the organization of perinatal health care and
formulation of renewed perinatal health care policy. A broad network of actors resulted in the
formulation of diverse measures. Policy emphasized preventive care and measures throughout
the full spectrum of the perinatal period: from preconception health through to and including the
puerperium. It was acknowledged that perinatal health is not solely influenced by biological factors
but by social and environmental factors as well and that perinatal health affects health outcomes
in adult life.23 This resulted in the policy that intervention in perinatal mortality requires municipal
involvement. The policy process occurred in a relatively short period. The most important process
factors where the nature of the topic and the fact that perinatal mortality rates and the public
profile of the debate. This promoted that a broad scope of professionals was engaged in the policy
process. In contrast, prior to the debate, only policy makers of the department of curative health
were involved. Important contextual factors were the organization of perinatal health within the
tiered health care system and divergent views amongst perinatal health care providers.

Key elements in the agenda setting

Firstly, the topic could be targeted because the perinatal mortality problem had been quantified by
PERISTAT and by additional research. It was this quantification of perinatal health data that created
urgency to act amongst politicians, Aldermen, and the preventive sector. Prior to the debate, the
organization of perinatal health had not been evaluated. The system was deemed to be infallible by
the majority of policy makers (and society).
Secondly, the nature of the topic was engaging to all actors in the debate. The fact that the topic
of perinatal mortality concerns a relatively large group in society made this a subject of interest to
politicians, policy makers, health care professionals and the public. The media was eager to be an
intermediary and fueled the debate. Especially the involvement of politicians and the media set the
speed of the debate. Gynecologists and midwives could not agree upon the ideal organization of
perinatal care. However, the high public profile and the speed of the debate forced them to agree
that the perinatal health statistics required changing in the organization of perinatal healthcare.
Whilst the whole debate was fiery, there was agreement that rapid interventions were necessary
amongst all actors involved.
A key result of the agenda setting was the founding of a new network at a national policy level
that committed to identify solutions to intervene in perinatal health. Two actors in these networks
(or subnetworks in the perinatal mortality policy process) should be mentioned specially. Firstly

32
the Baby Club, that consisted of policy makers from different departments of the Health Ministry.
This network promoted that the department of public health became involved and that measures
reflected a more socioeconomic environment oriented approach. Secondly, the Health Ministry
appointed a Steering Group which promoted that interventions were selected after input from a
multidisciplinary range of actors.

Key elements in the formulation of policy

The relatively unfavorable perinatal mortality rates caused a broad multidisciplinary network to
recognize that change in the organization of perinatal health care was required. Prior to these
numbers, perinatal health policy was restricted to the department of curative health of the Health 2
Ministry. It can be said that the multidisciplinary scope of actors that arose during the agenda

A policy analysis of perinatal health care

setting was the foundation for the diversity of the contents of policy. Firstly there was a shift in actors
involved in policy making: where first policy was only made by the department of curative care, the
department of public health became an important actor in policy making. This was enabled by the
fact that budget was allocated to the department of public health. They had formulated that policy
should incorporate that more attention should go out to non-medical risk factors and that local
municipalities should be involved in effectuation of community based health care. The Perinatal
health issue became an icon project to effectuate this vision. The academic field became involved
in the selection and effectuation of interventions. Secondly, with regards to the key concept to
intervene throughout the whole range of perinatal chain from preconception care to care in the
postpartum period. The Steering Group should be mentioned. The Steering Group functioned as a
bridge between the research field (providing evidence to point out the rationale and the evidence
for interventions), midwives and gynecologists and the policy field. This promoted collaboration
and the acceptance of measures by the curative field.

Strengths and limitations

To our knowledge this is the first policy analysis aiming to summarize the policy process that
took flight after the EURO-PERISTAT reports. This limits comparison to what extent policy measures
to reduce perinatal mortality have been taken in other European countries and why they were
taken. This is one of the aims of EURO-PERISTAT.3
There are many frameworks for the evaluation of policy processes. The policy triangle provided
a suitable framework for our policy analysis. It was specifically designed to identify the multitude of
factors (content, process, context and actors) that affected policy. In this model context and process
factors are equally as important as the actors. We did not investigate changes in the roles of actors
or stakeholders, because we deemed these roles as fairly consistent within the relative short period
of our study. Therefore this study is of limited value to identify stakeholders for future advocacy of
policy. In order to identify stakeholders for future advocacy we would recommend a prospective
policy analysis.
Strength of this policy analysis is the triangulation of methods, which provided the opportunity
to cross-verify findings from different sources. With the brief stakeholder analysis and snow-ball

33
sampling we aimed to identify key informants for interviews. Triangulation of data from these data
sources provided grounds for a coherent policy analysis. However, while triangulation provides
the opportunity to verify data, it does not exclude subjective interpretation. The authors of this
manuscript are active in the perinatal healthcare field and were involved in the Healthy Pregnancy
4 All project, one of the initiatives that came out of the policy process evaluated in this article. As
researchers within the field we noticed policy changes and new bridges amongst professionals
in the perinatal health care field, which provided grounds to conduct this study. However, being
members of the perinatal health care field can also introduce subjectivity in interpretation of
findings. We attempted to limit any potential bias by having the first authors (who were not
involved in any of the events prior to 2011) conduct the analysis and the other two authors verify
findings. To avoid bias in observations it would have been ideal to verify findings with an external
observer, without any involvement in the policy process. However, to our knowledge everybody
with enough authority to verify findings, would by definition have a role in the perinatal field and
thus per definition have a potential bias due to their position.
Many countries have highly specialized obstetric care systems and underdeveloped
collaboration between curative and public health care like the Netherlands.24 We believe this study
can generate thought regarding contents of policy, as it is largely based on current literature (e.g.
importance of socioeconomic determinants in health), which is applicable regardless of system
factors.

Practical implications and recommendations

The implications and recommendations for health care professionals and policy makers confronted
with health issues in a similar fragmented field can be summarized as follows:
• Demonstrating the importance of the problem (numbers are essential) can help to bring policy
issues to the agenda.
• Placing the problem in a multidisciplinary context can result in identification of new solutions.
Collaboration between the academic field (knowledge) and politics (money and policy), and
between the curative and preventive sector resulted in new measures. This is an example of
how investing time in the identification of the stakeholders with whom you share a problem
can be rewarded with better collaboration in the selection of interventions.
• Urgency and fast actions can be enforced by engaging the political field and the media.
However, this should be done with caution as it can polarize discussions in such a way that they
may become incontrollable.
• Finding a network that is aware of your problem or related problems can increase the likelihood
that resources are allocated to solve your problem. In this policy process the perinatal mortality
debate proved to be a catalyst for solutions to related problems.
• Reducing perinatal mortality and inequalities in perinatal health requires integration of care
from the curative and the preventive sector. National governments need to collaborate with
municipalities to deliver perinatal health care that addresses socio-environmental determinants
in a tailored fashion.

34
CONCLUSIONS
Over the past decade politicians and policy makers have acknowledged that the high perinatal
mortality rates were a national health issue over the past decade. This resulted in new policy.
Regarding content of formulated policy, we observed that prior policy policies were related to care
within the curative setting. Key features of new policy were firstly that intervention was necessary
throughout the full range of perinatal care (from the preconception care period to the postnatal
period). Secondly, interventions would have to address socio-demographic factors that influence
perinatal health. This shift to addressing socio-environmental determinants of perinatal health
requires municipal involvement.
The broad range of actors led to the diversity of interventions. Where there was disagreement 2
regarding intervention selection at first, agreement and interventions selection was enforced by

A policy analysis of perinatal health care

political pressure and mediated by the Steering Group. However, according to politicians, future
debates should reveal the role of the perinatal healthcare field in further solutions of the problem:
“From now on the ball is in their court.” This policy analysis focused on formulated policy. Future
research needs to evaluate the extent to which policy has been implemented and been effective in
reducing perinatal mortality.

CONFLICTS OF INTERESTS
The last two authors participated in the study as respondents. The research team has received
funding from the Ministry of Health, Welfare and Sports in order to execute the Healthy Pregnancy
4 All study.

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sterdam]. Ned Tijdschr Geneeskd, 2011. 155: p. A3130.
9. De Vries, R., M. Nieuwenhuijze, and S.E. Buitendijk, What does it take to have a strong and independent profession of mid-
wifery? Lessons from the Netherlands. Midwifery, 2013. 29(10): p. 1122-8.
10. Christiaens, W., M.J. Nieuwenhuijze, and R. de Vries, Trends in the medicalisation of childbirth in Flanders and the Nether-
lands. Midwifery, 2013. 29(1): p. e1-8.

35
11. Walt, G. and Gilson L., Reforming the health sector in developing countries: the central role of policy analysis. Health Policy
Plan, 1994. 9(4): p. 353-70.
12. Buse, K., N. Mays, and G. Walt, Making health policy. First ed. Understanding Public Health, Open University Press London
2005.
13. Government of the Netherlands: Documents and publications Accessed on 16 January 2014]; Available from: http://www.
rijksoverheid.nl/ministeries/vws/documenten-en-publicaties/kamerstukken.
14. Varvasovszky, Z. and R. Brugha, A stakeholder analysis. Health Policy Plan, 2000. 15(3): p. 338-45.
15. Buitendijk, S.E. and J.G. Nijhuis, Hoge perinatale sterfte in Nederland in vergelijking tot de rest van Europa. [High perinatal
mortality in the Netherlands compared to the rest of Europe]. Ned Tijdschr Geneeskd, 2004. 148(38): p. 1855-60.
16. Timmermans, S., et al., Individual accumulation of heterogeneous risks explains perinatal inequalities within deprived
neighbourhoods. Eur J Epidemiol, 2011. 26(2): p. 165-80.
17. de Graaf, J.P., et al., Perinatale uitkomsten in de vier grootste steden en de prachtwijken in Nederland. [Perinatal outcomes
in the four largest cities and in deprived neighbourhoods in The Netherlands] Ned Tijdschr Geneeskd, 2008. 152(50): p.
2734-40.
18. Stuurgroep Zwangerschap en Geboorte: Een goed begin. [Advisory Committee on ‘Good care during pregnancy and child
birth’]. 2009; Available from: http://www.goedgeboren.nl/netwerk/Multimedia/Get/641.
19. Troe, E., et al., Ethnic differences in gestational duration, preterm and very preterm delivery. Submitted for publication,
2008.
20. Denktas, S., et al., An urban perinatal health programme of strategies to improve perinatal health. Matern Child Health J,
2012. 16(8): p. 1553-8.
21. van Voorst, S.F., et al., Effectiveness of general preconception care accompanied by a recruitment approach: protocol of a
community-based cohort study (the Healthy Pregnancy 4 All study). BMJ Open, 2015. 5(3): p. e006284.
22. Vos, A.A., et al., Effectiveness of score card-based antenatal risk selection, care pathways, and multidisciplinary consultation
in the Healthy Pregnancy 4 All study (HP4ALL): study protocol for a cluster randomized controlled trial. Trials, 2015. 16: p. 8.
23. Barker, D.J., The foetal and infant origins of inequalities in health in Britain. J Public Health Med, 1991. 13(2): p. 64-8.
24. Heath, C. and N. Staudenmayer, Coordination neglect: How lay theories of organizing complicate coordination in organiza-
tions. Research in Organizational Behavior, 2000. 22: p. 153-192.
25. Leichter, H., State health policy analysis: on the abuse of metaphor and the pathology of variation. J Health Polit Policy Law,
1997. 22(3): p. 897-906.
26. Schafer, W., et al., The Netherlands: health system review. Health Syst Transit, 2010. 12(1): p. v-xxvii, 1-228.

36
2

A policy analysis of perinatal health care

37
SUPPLEMENTARY BOX 1: The catalyzing effect of the perinatal

mortality debate: additional events in the perinatal health field

• Research field
- setting of a research agenda (Signalement study)
- calling attention for more funding for research in perinatal
healthcare: appointment of the Netherlands organization for Health
Research and Development – Pregnancy and Childbirth program
- forming of research consortia
• Training
- accelerated training of more maternity nurses to compensate for
shortages
- expansion of the number of training places for midwives and to
professionalize the training
• Financial
- additional tariff for midwives in disadvantaged neighborhoods
- debate about personal contribution for outpatient deliveries
• Organizational
- founding of Birth centers
- debate about integrated care

38
SUPPLEMENTARY TABLE 1

This Table presents the results of the stakeholder analysis performed according to methods proposed by Varvaskovszky and
co-authors.17 This analysis was performed to assist the actual policy analysis in identifying key-informants and understanding
their roles in the policy process. Characteristics of each of these stakeholders were identified based on preknowledge before
the policy analysis and new insights as a result of triangulation of data from the document analysis and interviews. These char-
acteristics are: (1) their interest in perinatal mortality, (2) their influence, (3) their position (supportive, opposed, and neutral)
and (4) the impact of perinatal mortality on them.27

Stakeholders Involvement Interests Power / Position Impact of is-

Influence sue on actor

Erasmus Medical Center Provision of information regard-

- Department of Obstet- ing the health issue and potential
HIGH MEDIUM SUPPORTIVE LOW
2
rics and Gynaecology interventions and evaluation of

A policy analysis of perinatal health care

these interventions (from a clin-
ical and scientific point of view).

Ministry of Health, Wel- Accountancy regarding the HIGH HIGH SUPPORTIVE HIGH
fare and Sport Health system and signaling of
problems in the Health system,
inequalities - from a policy mak-
ers point of view.
Member of chamber Committing to decrease perina- HIGH HIGH SUPPORTIVE HIGH
tal health inequalities amongst
ethnic minorities from a political
- socialistic point of view.

Municipal Health Ser- Provision of experiences in identi- HIGH MED / LOW SUPPORTIVE MEDIUM
vice of Rotterdam fication of the need to intervene
in Perinatal health and to seek
collaboration between municipal
parties, academic hospitals and
community health care providers.

The Royal Dutch Organ- Accountancy regarding midwives HIGH HIGH / MED SUPPORTIVE HIGH
isation of Midwives and their practices (guidelines
and education) in the field and
advocacy of midwives.

Dutch Society of Ob- Accountancy regarding profes- HIGH HIGH / MED SUPPORTIVE MEDIUM
stetrics and Gynaecol- sionals and their practices (guide-
ogy lines and education) in the field
and advocacy of gynecologists.

Netherlands Organiza- Delivery of knowledge and eval- MEDIUM HIGH SUPPORTIVE LOW
tion for Applied Scientif- uation of interventions regarding
ic Research perinatal health.

Institute for public Delivery of knowledge and eval- MEDIUM HIGH NEUTRAL LOW
health and environment uation of interventions regarding
perinatal health.

39
THE DUTCH NATIONAL SUMMIT ON
PRECONCEPTION CARE:
A SUMMARY OF DEFINITIONS,
EVIDENCE AND RECOMMENDATIONS
S.Temel, S.F. van Voorst, L.C. Potjer, A.J.M Waelput, M.C. Cornel,
S.R. de Weerd, S. Denktaş E.A.P. Steegers
Jouurnal of Community Genetics 2015; 6(1):107-15
ABSTRACT
Objectives: Preconception care is a primary preventive approach in which prepregnancy risk factors
are addressed in order to prevent adverse pregnancy outcomes. Although benefits of preconception
care are acknowledged, consensus on concepts of preconception care and approaches for
implementation in the Netherlands are currently lacking. Due to the comprehensiveness and
multidisciplinary nature of preconception care consensus could be a prerequisite to develop or
implement approaches to deliver preconception care.
Methods: A literature-based consensus meeting was organized to achieve consensus about (I) the
definition, (II) categories, (III) target groups, (IV) prepregnancy risk factors and interventions and (V)
risk assessment instruments.
Discussion: Preconception care is only delivered in a small scale and consensus on the content
and how the care should be delivered is not well documented. Consensus regarding the content
and delivery of preconception care is necessary to upscale preconception care and to commit both
curative and public health in their responsibility in preconception care.
Conclusions: This paper presents a summary of the reached consensus and the identified
knowledge gaps during the meetings.

42
INTRODUCTION
Preconception care (PCC) is widely recognized as a way to optimize women’s health through
biomedical and behavioural change prior to conception, ultimately to improve pregnancy
outcomes. In terms of prevention, PCC is primary prevention for the future baby and secondary
prevention for prospective mothers. When these appropriate secondary and primary preventive
measures are taken public health benefits are achievable by prevention and treatment of identified
risk factors (e.g. smoking, alcohol abuse, obesity and infectious diseases) and improvement of
perinatal health potentially leading to improvement of health later in life.
Despite recognition of the importance of PCC in the Netherlands within curative care and
governmental policy makers,1 PCC is still only delivered on a small scale and not in a uniform
manner. Lack of consensus regarding the content and the delivery of the care seems to be an
underlying cause. This consensus is important to provide caregivers with a foundation for further 3
implementation of PCC. Consensus is also a necessary first step in creating of awareness among

A national summit on preconception care

caregivers regarding their societal responsibilities in primary and secondary prevention. Therefore,
a consensus meeting was organized to identify gaps and essential targets to contribute to policy
thinking for implementation of PCC. Point of departure was a comprehensive literature study. This
paper summarizes results of the meetings. These results can be used to create commitment and
responsibility amongst curative care givers and public health policy makers to keep the debate
going in the content of PCC.

METHODS
A comprehensive literature study was performed to provide a starting point to address five core
subjects: 1) the definition of PCC, 2) categories of PCC, 3) relevant target groups and methods for
outreach, 4) risk factors which should be taken up in PCC (an evidence update as of 2008) and
effective interventions (evidence as of inception of databases), and 5) risk assessment instruments.
Despite increasing evidence of paternal influence on pregnancy outcome and the crucial influence
of men on their partners’ health behaviours, this meeting – and therefore the literature study -
firstly focussed on PCC for women.2-4 This meeting does not have its focus specifically on lifestyle
risk factors, however we would like to point out that we recently have published another systematic
review regarding effectiveness of PCC interventions on lifestyle risk factors in the Preconception
Phase.5 The meeting, organised by the Erasmus MC in Rotterdam, consisted of two one day sessions
(January 2012 and April 2012). Propositions for consensus – based on the literature - were presented
as a starting point for the discussion. Participants included:
• Care givers (midwives, general practitioners, gynaecologists, clinical geneticists, an occupational
health physician);
• Representatives from professional organizations of the care givers (Regional Organisational
Support for Primary health care [ROS]);
• Governmental representatives (the Ministry of Health Welfare and Sport, the Commission
for Perinatal Health [College Perinatale Zorg], a Municipal Health Service [GGD Rotterdam-
Rijnmond]);

43
• Health insurance companies and the Health Care Insurance Board;
• Funders of scientific research (the Netherlands Organisation for Health Research and
Development [Zon MW]);
• Providers of health care expertise (the Health Council of the Netherlands [Gezondheidsraad],
the National Institute for Public Health and the Environment [RIVM], the Dutch National Genetic
Resource and Information Center [Erfocentrum], the Dutch Foundation of Preconception Care,
Organisation for Applied Scientific Research [TNO], the Dutch birth registry (Netherlands
Perinatal Registry [PRN]);
• Patient-consumer federation (the Dutch Genetic Alliance of Parent and Patient organizations
[VSOP]);
• Other relevant disciplines (department of medical ethics, epidemiology).
• Sessions were chaired by independent experts on PCC. Achieved consensus, lack of consensus
and knowledge gaps were recorded. These records were verified by participants after each
session.

EXPERTS DISCUSSION
Results will be presented per core subject in a fixed format: an introduction, the proposal, achieved
consensus (in case of agreement), lack of consensus (if any) and identified knowledge gaps
resulting in recommendations for future research.

I. DEFINITION OF PCC
Introduction
Various definitions for PCC have been formulated. The definition is an important take off point in the
debate around the content of PCC. In 1992 the following definition was included in PubMed’s Mesh
database: “An organized and comprehensive program of health care that identifies and reduces
a woman’s reproductive risks before conception through risk assessment, health promotion, and
interventions.6 In 2005 the Centers for Disease Control and Prevention (CDC) and the March of
Dimes recognized the need to state that PCC is a continuum of care throughout the various stages
of the reproductive life of women. This was incorporated in their definition: “A set of interventions
that aim to identify and modify biomedical, behavioural, and social risks to a woman’s health or
pregnancy outcome through prevention and management, emphasizing those factors that must
be acted on before conception or early in pregnancy to have maximal impact.”7 In 2007 the Health
Council of the Netherlands presented a definition in line with the CDC: “Preconception care is the
entire range of measures designed to promote the health of the expectant mother and her child,
which, in order to be effective, must preferably be adopted prior to conception.” 1

Proposition
To adapt the definition of the CDC and the March of Dimes, due to the different elements of risk
factors, defined outcomes and the defined timeframe.

44
Consensus
• There was agreement with the proposition.
• To add that: PCC should be regarded as a programme and that PCC includes psychosocial risks,
non-medical risks (e.g. financial problems and domestic violence) counselling and informed
decision making.
• To replace ‘woman’s health’ with ‘parental health’.
• To replace ‘pregnancy outcome’ with ‘the health of their future child’, prolonging the timeframe
targeted by PCC.
• A note should be added to the definition about the potential of PCC to reduce perinatal
mortality and morbidity.
• The consensus meeting resulted in the following definition: “A set of interventions and/or
programmes that aims to identify and enable informed decision-making to modify biomedical, 3
behavioural, and (psycho)social risks to parental health and the health of their future child,

A national summit on preconception care

through counselling, prevention and management, emphasizing those factors that must be
acted on before conception and in early pregnancy, to have maximal impact and/or choice.
• Preconception care may be a good opportunity to reduce perinatal mortality and morbidity.

Knowledge gaps/recommended future research

• Although major steps are to be made in the implementation of PCC for women first, it is
desirable to achieve consensus on PCC for men, in the future.
• Perinatal mortality and morbidity is a more important outcome for policy makers. Therefore
trials should also address pregnancy outcomes (besides behavioural change) as an outcome
measure of the effectiveness of PCC (see Figure I).

II. CATEGORIES OF PCC

Introduction
PCC is meant to improve the health of mother and child in various ways. The Dutch Health Council
provides the following categorization of methods for PCC delivery:
• Collective measures are aimed at the general population to improve preconception health. An
example is campaigns on the use of folic acid.
• General individual PCC is detection and management or intervention on risk factors, in couples
planning a pregnancy within the general population. The general nature resides from the fact
that these couples mostly do not have a known or predefined preconception risk(profile).
• Specialist individual PCC is provided for a) couples with a known or predefined risk for an
adverse pregnancy outcome (e.g. Diabetes) or b) couples who are referred from general
individual PCC after risk assessment (e.g. when diabetes is detected).
• Recognition of the different forms of PCC is important in the implementation of PCC.
Categorization provides a basis to identify professionals with core responsibilities in a category,
to tailor a feasible recruitment approach and applicable target group.

45
 H3 F1
FIGURE 1: Outcomes of PCC
Preconception risk factors potentially and behavioural change may influence foeto-maternal health throughout the pericon-
ception period, pregnancy as well as during childhood and adulthood. Health during reproductive age will subsequently affect
the outcomes of subsequent pregnancies and the health of future generations.

Childhood Growth development and health

& adulthood of the child and individual later in
life

Pregnancy Perinatal health outcome

Behavioural change and maintenance

Effects on embryonal/ foetal

development and health
of PCC risk factors

Prior to Preconception health

conception

Individual PCC Collective PCC

Proposition
• Not to change the categorization of PCC.
• Addition of care pathways to the elements of PCC. They can facilitate implementation of
individual PCC in a uniform and locally tailored manner. Care pathways are a means of
achieving multidisciplinary agreements on organization and efficient shared care. They should
be evidence based and in line with local guidelines and available care facilities.8

Consensus
• Care pathways were recognized to be valuable, specifically where they address socio-medical
risk factors. Professional organizations should have a leading role in the development in care
pathways, specifically to achieve multidisciplinary agreements.

46
Disagreement
• There is unclarity regarding which health care professional has a core responsibility in which
category. The line between general individual PCC and specialized PCC is not very evident.
There are caregivers that could address both general and specialized individual PCC. The
difficulty lays in the education and/experience in addressing specialistic risk factors. As PCC has
a very broad content; it seems merely impossible for one caregiver to address all risk factors.

Knowledge gaps/recommended future research

• There is a need to define the role of different professions within the Dutch Healthcare system
within different categories of PCC. The collaboration between public and curative health, and
delegation of tasks (e.g. to qualified physician assistants or nurses) should be explored further.
This task can be fulfilled by the Commission for Perinatal Health (CPZ) which has now appointed 3
a committee that will develop a consensus based multidisciplinary guideline. This guideline will

A national summit on preconception care

explicate specific roles of health care workers.

III. REACHING TARGET GROUPS

Introduction
So far, no (inter)national consensus exists as to whom PCC should be offered. The target population
can be divided into four major groups: (1) the general population, (2) all men and women of
reproductive age (3) men and women aiming to conceive and (4) men and women with predefined
high risk groups (e.g. due to previous pregnancy complications, genetic risks, chronic illness or
medication use).
Reaching women and men before the onset of pregnancy is crucial for effective PCC. Women
neither actively seek PCC consultation, nor do they accept the offer to attend a consultation.9 In
every day practice clinicians do not often initiate a PCC consultation, nor do they recommend
it to women.10,11 The curative setting and the public health setting in contact with women of
childbearing age should be aware of the importance of preconception health promotion. However
there is a lack of awareness or perhaps sense of responsibility under these professionals about their
responsibility and potential role in preconception health promotion.
Research on why the outreach of PCC is limited and how this short-coming can be addressed,
is scarce. Several studies have indicated that an important problem with reaching parents to be
on time is that many women do not plan pregnancies.12,13 Another challenge is adapting the
PCC approach to reach specific target groups. The importance is recognized by trials evaluating
outreaches of PCC programs.14-16 Above all, research on effective (tailored) methods to reach target
groups for PCC are lacking.17,18

47
Proposition
• Increased awareness and specification of their role in executing PCC should be contemplated by
the following care givers/ organizations: governmental organisations, care providers (midwives,
general practitioners, pediatricians, gynecologists and medical specialists in general), youth
and family centers, peer educators, social welfare services, and schools.

Consensus
• There was consensus on the fact that preconception health promotion needs broad support
from actors with different relations to the target group. The following actors in general were
identified additional to the professionals above, either as a direct link to the target population
or as a medium: municipal health service, paramedics (e.g. dieticians and dentists), pharmacists,
occupational health physicians, all health promotional institutes in general that address people
of childbearing age, institutes focusing on migrants, the social network around future parents
(e.g. aunts, grandmothers), policy makers and means of communication (e.g. internet). The
need for involvement and collaboration of curative health professionals and public health
professionals is therefore acknowledged.
• Tailored approaches should be applied by actors for the different target groups of PCC.
Specifically teenagers are a group of interest because early sensitisation could promote timely
behavioural change or utilisation of PCC services later in life.

FIGURE 2: Target approach to reach women with

Target group:
Diabetes Mellitus
Women with Diabetes Mellitus
This figure shows a potential approach to improve precon-
ception health and to target women with Diabetes Melli- Goal: To increase awareness
tus to utilize Preconception Care when they contemplate Education on PCC as part of reproductive
health education.
pregnancy later in life.

Goal: Recruitment towards PCC

When periodic monitoring of Diabetes Mellitus
is conducted by a physician or medical
specialist the remark to come back for PCC
when she contemplates pregnancy.

Goal: Provision of PCC

Individual risk assessment and adjusting
treatment when she contemplates pregnancy.

48
Knowledge gaps/recommended future research
• The consensus meeting concentrated on identifying actors to enlarge awareness and outreach
of preconception health promotion amongst target groups. Feasible approaches should be
developed per actor; per target group.
• There is a gap in practice as to how the above mentioned actors optimally could have a role.
A potential schematic approach to reach women with Diabetes Mellitus is shown in Figure. 2.

IV. RISK FACTORS AND INTERVENTIONS AS PART OF PCC

Introduction
For the delivery of PCC there has to be consensus on the content. This should be based on known 3
risk factors for adverse pregnancy outcomes and effective interventions to address them in the

A national summit on preconception care

preconception period. A risk factor- and intervention review was conducted to form a basis for the
discussions.

Risk factors
A review by Jack et al., was conducted in 2008 to provide evidence for risk factors to be taken up
in PCC.19 To update this review for the consensus meeting a search was conducted in PubMed, as
of 2008. Selection was performed according to predefined criteria: the study assesses risk factor(s)
which are present in the preconception period and the study reports an association with an
adverse pregnancy outcome. Three reviewers independently assessed eligibility and performed
data extraction. The search resulted in 2214 articles of which 178 articles were included.

Interventions
A systematic search was conducted to assess efficacy of available PCC interventions in PubMed,
Embase and Web of Science from 1900 to January 2012. Selection was performed according to
predefined criteria: the study assesses interventions, addressed in the preconception phase for an
adverse pregnancy outcome. Two reviewers independently assessed eligibility of 678 articles and
performed data extraction on 104 included articles.
Table 1 gives an update of the quality of the evidence for the risk factors per domain with
interventions where available. Strength of evidence was assessed according to the Canadian Task
Force on Preventive Health Care.20

Proposition
• Identified risk factors and available interventions with a level of evidence of I-A to II-3 should be
included as part of evidence based PCC.
• Identified risk factors with a level of evidence of I-A to II-3, but without evidence based
interventions, should be prioritized for development of interventions.

49
TABLE 1: Q
 uality of the evidence for preconception risk factors and interventions to improve

maternal and/or infant health and consensus on uptake in PCC

Risk domain Risk factors Outcome Intervention Consensus

Health care Interpregnancy intervals
II-2 +
promotion (<6 months and >60 months)
Lack of physical exercise II-2 I-a +
Unplanned pregnancy III +
Immunizations Human Papilloma Virus (HPV) II-2 -
MMR II-3 II-2 +
Hepatitis B III -
Varicella III +
Influenza III -
DTP III +
Infection Syphilis I-a +
HIV I-b +
Periodontal disease I-b +
Bacterial vaginosis I-b +
Asymptomatic bacteriuria II-1 I-a +
Herpes Simplex Virus (HSV) II-1 +
Chlamydia II-2 +
Toxoplasmosis II-2 +
GBS II-2 +
Tuberculosis II-2 +
Hepatitis C III +
Cytomegalovirus (CMV) III +
Parvovirus III +
Malaria III +
Gonorrhoea III +
Chronic medical Diabetes mellitus type 1 or 2 I-a I-a +
conditions Thyroid disease II-1 +
Phenylketonuria (PKU) II-1 +
Seizure disorders II-2 +
Hypertension II-2 +
Systemic Lupus Erytromatosus (SLE) II-2 +
Chronic renal disease II-2 +
Cardiovascular disease II-2/II-3 +
Thrombophilia II-3 +
Asthma II-3 +
Rheumatoid arthritis (RA) III +
Psychiatric Depression and anxiety disorders II-2 +
conditions Bipolar disorder II-2 +
Schizophrenia II-2 +
Maternal Alcohol I-a I-a +
exposure Tobacco I-a +
Illicit substances II-2 +

50
TABLE 1 continued

Risk domain Risk factors Outcome Intervention Consensus

Genetic risks Genetic disorder(s) or carrier ship

II-2 +
in one of the prospective parents
Ethnicity based risks II-3 +
Positive family history II-3 +
Recurrent miscarriages III II-2
Known genetic conditions II-3
Nutrition Inadequate folate intake I-a I-a +
BMI > 30 kg/m2 I-b I-a +
BMI < 18 kg/m2 II-2 +
Insufficient vitamin B12 II-1 + 3
Inadequate dietary intake II-2 I-a +
Western Dietary pattern II-2 +

A national summit on preconception care

Excessive vitamin E intake II-2 +
Insufficient Vitamin D II-3 +
Insufficient or excessive vitamin A intake III +
Eating disorders III +
Environmental Occupational exposure
II-2 +
exposures (e.g. chemicals, solvents)
Household exposures
III +
(e.g. PCB’s, solvents, metals (lead))
Psychosocial Inadequate financial resources II-2 +
stressors Interpersonal violence II-2 +
Medication Prescribed medication II-1 +
Herbs / herbal products /
II-1 +
weight loss products
Over the counter drugs III +
Reproductive Prior preterm birth I-a +
history Prior miscarriage I-a +
Prior fetal growth restriction II-2 +
Prior caesarean delivery II-2 +
Prior stillbirth II-2 +
Uterine anomalies II-3 +
Special groups Immigrant and refugee populations II-2
Women who survived cancer II-2
Women with disabilities III

*Quality of evidence
I-a: at least 1 properly conducted randomized controlled trial BEFORE pregnancy
I-b: at least 1 properly conducted randomized controlled trial not necessarily before pregnancy
II-1: well-designed controlled trials without randomization
II-2: cohort or case-control studies
II-3: multiple time series with or without intervention or dramatic results in uncontrolled experiments
III: opinions: clinical experience, descriptive statistics, case reports or reports of experts committees

51
Consensus
• Table 1 shows the consensus achieved per risk factor, regarding the uptake as part of PCC.
• There were remarks considering the uptake of the following risk factors in PCC:
·· Group B streptococcus (GBS): Due to the recurrence of GBS colonisation after treatment,
it is not considered beneficial to screen all women preconceptionally for GBS. However,
PCC can identify women with previous GBS infection or neonatal complications due to GBS
colonization. For these women a management plan for their pregnancy and delivery can be
formulated.
·· HPV immunization: Although HPV carrier status is common, fetomaternal transmission rates
and consequent neonatal outcomes are infrequent; there was consensus not to incorporate
HPV carrier detection and immunization in PCC.
·· Hepatitis B immunization: Where Hepatitis B infection is present in one of the future
parents, routine clinical care was thought to be sufficient together with the local policy in
pregnancy regarding vaccination of the neonate after birth.
• Although the review did not point out the following risk factors; the experts noted the following
risk factors to be taken up as part of PCC:
·· Chronic medical conditions: such as inflammatory bowel disease (Colitis, Crohn’s disease),
women with organ transplants, previous thrombotic event or embolism.
·· Genetic risks: consanguinity.
·· Exposures: Occupational exposure to working shifts and stress, sauna, diving and passive
smoking as part of household exposure.
·· Psychosocial stressors: adverse childhood events.
·· Reproductive history: subfertility, prior pregnancy complications, prior congenital
anomalies, prior neonatal complications and advanced maternal age (defined as older than
36 years).
·· The discussion about risks that should be addressed by the PCC provider set aside,
prospective parents may have questions (e.g. with regards to fertility and sexual health).
PCC providers should assess needs and inform or refer where necessary.
• The current proposal focuses on individual risk factors. The participants agreed that the
effect of risk accumulation should be recognized. Risk accumulation is the phenomenon that
combinations of risk factors augment the total risk of the individual to a larger extent than the
sum of the individual risks.21

Knowledge gaps/recommended future research

• More research is needed regarding the Population Attributable Risks (PAR) of preconception
risk factors and combined effects of risk factors.
• A remark can be made by the risk factor ‘unplanned pregnancy.’ It is unclear whether this
considers unplanned pregnancies that are welcome or not welcome. This might be an
important factor affecting pregnancy outcome as risky behaviour is more likely to happen

52
 when a pregnancy is unwanted. More insight is necessary in the contributory risk component in
unplanned pregnancy: unwantedness versus the unplanned nature.
• As the possible content of PCC is growing, there is a need for prioritization in the interventions
for a woman’s specific risk profile. There is no method to identify the best core of action and a
fixed format is not feasible due to inter-individual differences. PCC providers are subjected to
‘common sense’ in the prioritization of risk factors. This should be based on the impact of risk
factors and the feasibility of interventions.

V. RISK ASSESSMENT INSTRUMENTS

Introduction
Assessing preconception risk factors within all domains is time consuming and to stimulate a 3
uniform risk assessment; risk assessment instruments are necessary.

A national summit on preconception care

Available risk assessment instruments were identified:
• ZwangerWijzer is the most widely used instrument in the Netherlands.22,23 It is a validated
tool based on the Preconception Health Assessment form developed by Cefalo and Moos.24 It
is self-administered online questionnaire that assesses and informs about medical-, genetic-,
environmental- , occupational-, nutritional-, and lifestyle risk factors. The identified risks can
be emailed to a caregiver – to provide an agenda for individual PCC. A supportive program
provides the caregiver with a preconception patient record with protocols to address each
identified risk factor.25
• Slimmer Zwanger is a personal screening and coaching program provided by mobile phone
app.26 The application assesses nutrition and lifestyle behaviours by a self-administered
questionnaire. The application then provides motivational text messages and e-mail messages
to change risky behaviours. Effectiveness is currently being assessed.

Proposition
• To include generic risk assessment instruments suitable for the local setting in PCC.

Consensus
• Instruments with a wide range of detecting risk factors to limit the amount of questionnaires
are preferable.
• Risk assessment instruments can lead to awareness and therefore can function as an
intervention themselves.

Knowledge gaps/recommended future research

• Appropriate evidence-based standardised risk assessment instruments remain to be developed
or existing tools should be optimized (e.g. multilingual) and validated.

53
SUMMARY
In conclusion, consensus was achieved on the majority of the key elements of PCC, including the
definition, the categorisation, institutes and health care professionals which should play a role
in reaching target groups, the content and delivery and the need for development of evidence-
based risk assessment instruments. These elements give further insight in what should be
resolved in order to enlarge the scale at which PCC is delivered. Furthermore, these can be used
as starting points for policy makers and other relevant actors that take responsibility to develop
implementation strategies for PCC.
In order to develop a tailored PCC program, the needs of specific populations should be known
and resources should be in line with setting specific characteristics.
This consensus paper is based on current evidence. Biannual update on the evidence of
preconception risk factors and management is recommended to keep the debate going. This
debate is necessary to hold the commitment amongst the broad scope of professionals in the
curative setting and the public health care setting to collaborate regarding PCC.

CONFLICTS OF INTEREST
The last two authors participated in the study as respondents. The research team has received
funding from the Ministry of Health, Welfare and Sports in order to execute the Healthy Pregnancy
4 All study.

ACKNOWLEDGEMENTS
The Erasmus MC group is grateful to the following participants (in alphabetic order): I. Aalhuizen,
Royal Dutch Organization of Midwives (KNOV), Utrecht; A. Annegarn, Regional Organisational
Support for Primary health care (ROS), Amsterdam; R. Bakker, Erasmus University Medical Center,
Rotterdam; M. Beentjes, Royal Dutch Organization of Midwives (KNOV), Utrecht; M. Breden,
Netherlands Organisation for Health Research and Development (Zon MW), The Hague; G.J. Bonsel,
Erasmus University Medical Center, Rotterdam; T. Brand, VU University Medical Center, Amsterdam;
M.C. Cornel, VU University Medical Center, Amsterdam (chair); M.R. Crone, Leiden University
Medical Center, Leiden; J. Derksen, the Health Care Insurance Board, Diemen; S.B. Detmar,
Organisation for Applied Scientific Research (TNO), Child Health, Leiden; J. Dijs-Elsinga, Perinatal
Registry Netherlands (PRN), Utrecht; K. Dolsma, Dutch National Genetic Resource and Information
Center (Erfocentrum), Woerden; C. Hoving, Maastricht University Medical Center, Maastricht; A.A.J.
van Iersel, Ministry of Health Welfare and Sport, The Hague; S. Jans, Royal Dutch Organization of
Midwives (KNOV), Utrecht; A.H.J. Jaspar, National Association of General Practitioners (NHG),
Utrecht; L.P. ten Kate, Leiden University Medical Center, Leiden, Foundation of Preconception Care,
Soest; C. Kirpestein, IRIS, Tiel; A. Kruger-Adriaenssen, Agis, Amersfoort; P. Lakeman, VU University
Medical Center, Amsterdam; R. Lamara, Dutch National Genetic Resource and Information Center
(Erfocentrum), Woerden; A. Meeuwese, National Institute for Public Health and the Environment
(RIVM), Bilthoven; C. Oosterwijk, Dutch Genetic Alliance of Parent and Patient organizations

54
(VSOP), Soest; K. van der Pal- de Bruin, Organisation for Applied Scientific Research (TNO), Child
Health, Leiden; M. Prins, Ministry of Health Welfare and Sport, The Hague; S Rombout- de Weerd,
Albert Schweitzer Hospital, Dordrecht (chair); M. Rooseboom, Netherlands Organisation for Health
Research and Development (Zon MW), The Hague; V.W.T. Ruiz- van Haperen, Health Council of the
Netherlands (Gezondheidsraad), The Hague; K. Scheele, CZ, Tilburg; D.J. de Smit, MediClara Projects
BV, Abcoude; R.P.M. Steegers-Theunissen, Erasmus University Medical Center, Rotterdam; S. Veen,
Leiden University Medical Center, Leiden; P. Verloove- van Hoorick, Leiden University Medical
Center, Leiden; M. Vesters, Netherlands Organisation for Health Research and Development (Zon
MW), The Hague; E.A.P. van Vliet-Lachotzki, Dutch National Genetic Resource and Information
Center (Erfocentrum), Woerden; A.J.J. Voorham, Municipality of Rotterdam (GGD Rotterdam-
Rijnmond), University of Applied Sciences of Rotterdam, Rotterdam; A.J. Waarlo, University Medical
Center Utrecht, Utrecht; G. de Wert, Maastricht University Medical Center, Maastricht; S. Westerik, 3
Regional Organisational Support for Primary health care (ROS), Friesland; B. Wijsen, Commission for

A national summit on preconception care

Perinatal Health (CPZ), Utrecht; the Netherlands.

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1. Health Council of the Netherlands: Preconception care: a good beginning Health Council of the Netherlands. The Hague
2007. publication no. 2007/19E, ISBN 978-90-5549-657-0.
2. Frey KA, Navarro SM, Kotelchuck M, Lu MC. The clinical content of preconception care: preconception care for men. Ameri-
can journal of obstetrics and gynecology. Dec 2008;199(6 Suppl 2):S389-395.
3. Gage JD, Everett KD, Bullock L. A review of research literature addressing male partners and smoking during pregnancy.
Journal of obstetric, gynecologic, and neonatal nursing : JOGNN / NAACOG. Nov-Dec 2007;36(6):574-580.
4. Sternberg P, Hubley J. Evaluating men’s involvement as a strategy in sexual and reproductive health promotion. Health
promotion international. Sep 2004;19(3):389-396.
5. Temel S, van Voorst SF, de Jong-Potjer LC, et al. The Dutch national summit on preconception care: a summary of defini-
tions, evidence and recommendations. Journal of community genetics. Jan 2015;6(1):107-115.
6. Pubmed database. MeSH term Preconception care (Unique ID: D016742). http://www.ncbi.nlm.nih.gov/mesh/?term=pre-
conception+care, 2014.
7. Posner SF, Johnson K, Parker C, Atrash H, Biermann J. The national summit on preconception care: a summary of concepts
and recommendations. Matern Child Health J. Sep 2006;10(5 Suppl):S197-205.
8. Posthumus AG, Scholmerich VL, Waelput AJ, et al. Bridging between professionals in perinatal care: towards shared care in
the Netherlands. Matern Child Health J. Dec 2013;17(10):1981-1989.
9. Elsinga J, van der Pal-de Bruin K, le Cessie S, de Jong-Potjer L, Verloove-Vanhorick S, Assendelft W. Preconception counsel-
ling initiated by general practitioners in the Netherlands: reaching couples contemplating pregnancy [ISRCTN53942912].
BMC Fam Pract. 2006;7:41.
10. Gaytant MA, Cikot RJ, Braspenning JC, Grol RP, Merkus JM, Steegers EA. [Preconception counseling in family practice; a
survey of 100 family physicians]. Ned Tijdschr Geneeskd. May 23 1998;142(21):1206-1210.
11. Williams JL, Abelman SM, Fassett EM, et al. Health care provider knowledge and practices regarding folic acid, United
States, 2002-2003. Matern Child Health J. Sep 2006;10(5 Suppl):S67-72.
12. Delvoye P, Guillaume C, Collard S, Nardella T, Hannecart V, Mauroy MC. Preconception health promotion: analysis of means
and constraints. Eur J Contracept Reprod Health Care. Aug 2009;14(4):307-316.
13. van der Zee B, de Beaufort I, Temel S, de Wert G, Denktas S, Steegers E. Preconception care: an essential preventive strategy
to improve children’s and women’s health. J Public Health Policy. Aug 2011;32(3):367-379.
14. Denktas S, Bonsel GJ, Van der Weg EJ, et al. An urban perinatal health programme of strategies to improve perinatal health.

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 Matern Child Health J. Nov 2012;16(8):1553-1558.
15. Korenbrot CC, Steinberg A, Bender C, Newberry S. Preconception care: a systematic review. Matern Child Health J. Jun
2002;6(2):75-88.
16. Owens MD, Kieffer EC, Chowdhury FM. Preconception care and women with or at risk for diabetes: implications for commu-
nity intervention. Matern Child Health J. Sep 2006;10(5 Suppl):S137-141.
17. Bonsel GJ, Birnie E, Denktas¸ S, Poeran J, EAP S. Dutch report: Lijnen in de Perinatale Sterfte, Signalementstudie Zwanger-
schap en Geboorte 2010 . Rotterdam: Erasmus MC; 2010. Available at: www.nvk.nl/Nieuws/Dossiers/Dossier Perinatalezorg.
aspx. Retrieved January 5, 2011.
18. Janz NK, Herman WH, Becker MP, et al. Diabetes and pregnancy. Factors associated with seeking pre-conception care. Dia-
betes care. Feb 1995;18(2):157-165.
19. Jack BW, Atrash H, Coonrod DV, Moos MK, O’Donnell J, Johnson K. The clinical content of preconception care: an overview
and preparation of this supplement. American journal of obstetrics and gynecology. Dec 2008;199(6 Suppl 2):S266-279.
20. US Preventive Services Task Force. Guide to clinical preventive services. Baltimore: Williams and Wilkins; 1996.
21. Timmermans S, Bonsel GJ, Steegers-Theunissen RP, et al. Individual accumulation of heterogeneous risks explains perinatal
inequalities within deprived neighbourhoods. Eur J Epidemiol. Feb 2011;26(2):165-180.
22. Landkroon AP, de Weerd S, van Vliet-Lachotzki E, Steegers EA. Validation of an internet questionnaire for risk assessment in
preconception care. Public Health Genomics. 2010;13(2):89-94.
23. Stichting Erfocentrum en Erasmus MC. Preconception questionnaire [ZwangerWijzer]. www.zwangerwijzer.nl.
24. Cefalo R.C., Moos M.K. Preconceptional Health Care: A Practical Guide. St. Louis: Mosby-Year Book; 1995.
25. Preconception consultation tool for preconception care providers [Preconceptiewijzer]. www.preconceptiewijzer.nl.
26. Preconception consultation tool for prospective parents [Slimerzwanger]. www.slimmerzwanger.nl.

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57
EVIDENCE-BASED
PRECONCEPTIONAL LIFESTYLE
INTERVENTIONS
S. Temel, S.F. van Voorst, B.W. Jack, S. Denktaş, E.A.P. Steegers
Epidemiologic reviews 2014; 36(1):19-30
ABSTRACT
Objectives: Although the evidence for the associations between preconceptional risk factors and
adverse pregnancy outcomes is extensive, the effectiveness of preconceptional interventions to
reduce risk factors and to improve pregnancy outcomes remains partly unclear. The objective of
this review is to summarize the available effectiveness of lifestyle interventions prior to pregnancy
for women in terms of behavior change and pregnancy outcome.
Methods: A predefined search strategy was applied in electronic databases, and citation tracking
was performed. Study selection was performed by 2 independent reviewers according to predefined
criteria for eligibility: The intervention was performed preconceptionally on women regarding
alcohol use, smoking, weight, diet/nutrition, physical activity, and folic acid status (fortification and
supplementation) to achieve behavior change and/or improve pregnancy outcome. Quality and
strength of evidence were assessed by 2 independent reviewers.
Results: A total of 4,604 potentially relevant records were identified, of which 44 records met the
inclusion criteria.
Conclusion: Overall, there is a relatively short list of core interventions for which there is substantial
evidence of effectiveness when applied in the preconception period.

60
INTRODUCTION
Worldwide efforts are made to reduce adverse pregnancy outcomes. As many women do not realize
they are pregnant until the fifth week of pregnancy—when essential fetal processes have already
commenced—the first antenatal visit is relatively late to address perinatal risk factors1. As these risk
factors can mostly be identified, managed, or treated when they are detected preconceptionally to
prevent or limit fetal exposure, preconception care (PCC) has been identified as a promising form of
care to improve pregnancy outcomes. 2, 3
PCC is defined as “a set of interventions that aim to identify and modify biomedical, behavioral,
and social risks to a woman’s health or pregnancy outcome through prevention and management,
emphasizing those factors that must be acted on before conception or early in pregnancy to have
maximal impact” .4
Effective PCC interventions could be an opportunity to improve pregnancy outcomes. Although
the amount of evidence for preconceptional risk factors associated with adverse pregnancy
outcomes is growing, PCC is still based largely on the assumption that elimination of the risk factor
4
will reduce the chances of adverse perinatal outcomes rather than on evidence for the effectiveness

Review of preconception lifestyle interventions

of the preconceptional interventions itself. Risk factors in PCC are very diverse, reflecting the diverse
pathophysiology in the periconceptional period. Risk factors, from both parents, can be of genetic,
environmental, or behavioral origin. Therefore, a broad approach in PCC is necessary to optimize
perinatal health. In many countries, preconceptional health assessment focuses on women with
predefined risk factors, such as diabetes. PCC is offered much less frequently to women in the
general population without previously identified risk factors. Assessment of the general lifestyle
and behavioral risks such as alcohol consumption, smoking, the use of drugs, and nutritional diet
and folic acid supplementation seems to be offered mostly to these women with predefined risk
factors. More evidence is needed regarding the effectiveness of interventions aimed at general
lifestyle risk factors that are applicable to a large proportion of the couples aiming to conceive. This
evidence would not only help women with predefined risks but also be a boost for implementation
of PCC for the general population.
Furthermore, evidence for preconceptional health interventions is necessary to embed PCC
as an available health service—for professionals and for couples wishing to conceive— among
the general population. Also, concrete evidence is necessary to motivate policy makers, insurers,
and health-care providers themselves. Although it is challenging to reach target groups for PCC,
PCC is regarded to be a very welcome health service by couples wishing to conceive5. In order to
address these general risk factors in PCC, evidence-based preconceptional interventions to reduce
or eliminate these general risk factors are needed. Besides a Cochrane review in 20096 restricted
to randomized controlled trials, no systematic review comprising observational studies has been
conducted to address preconceptional lifestyle interventions for women. A systematic review
including observational studies is deemed valuable as the majority and most prominent studies are
observational because of the behavior changes that are included in PCC.

61
 The objective of this review is to provide an up-to-date overview of the effectiveness of
predefined lifestyle interventions on behavior change and improved pregnancy outcomes among
preconceptional women in the general population.

MATERIALS AND METHODS

Search strategy
Studies were identified initially with an electronic search in the databases Medline, Embase, and
Web of Science from inception to March 2012, restricted to the following languages (English,
Dutch, German, French, and Spanish) and to humans. The electronic search encompassed keywords
referring to the preconceptional time period, health-care promotion or intervention, the mother/
father or couple, and predefined risk factors. The detailed search is available in the Appendix.
Furthermore, citations of identified reviews were screened for eligible records.

Study selection
The following criteria for eligibility were applied to select studies: 1) The study included any kind
of intervention (e.g., varying from individual consultation to group education sessions performed
preconceptionally) regardless of duration or amount of visits of preconceptional women; 2)
the intervention focused on health promotion or on modification of any of the following risk
factors: alcohol, smoking, weight, diet/ nutrition, physical activity, folic acid fortification, and
folic acid supplementation (in relation to anomalies other than neural tube defects); and 3)
reported outcome(s) were behaviour change and/or risk factor modification and/or pregnancy
outcome (e.g., miscarriages, birth defects, premature birth, birth weight, low birth weight and/or
small for gestational age, and perinatal deaths). Regarding birth defects, development of neural
tube defects was not regarded as an outcome for folic acid supplementation, as this is already
considered evidence based in numerous studies7. Although fertility is an important outcome
of preconceptional interventions, this was regarded as a subgroup of interventions and was not
included in this systematic review. Records were assessed for eligibility on the basis of title and
abstract. The full manuscripts of these abstracts and of potentially relevant articles identified
with citation tracking were then evaluated to determine whether inclusion criteria were met.
Additionally, identified reviews were screened for potentially relevant references. Study selection
was performed independently by 2 reviewers (ST and SVV) with a third reviewer (SD) for
adjudication of discrepancies.

Data extraction
Predefined characteristics that were extracted were title; author(s); aim; intervention (how, when,
and by whom) per group (if applicable); study design; inclusion and exclusion criteria; participant
recruitment (time period of study, country, recruitment site, patient sampling method if specified);
methods of randomization/case or control selection/matching if applicable; data collection/
follow-up ( prospectively or retrospectively, sources of data, method and timeframe of assessment,

62
and blinding when specified); flowchart of participants; loss to follow-up (number and reasons
stated); baseline characteristics of the study population; setting of the intervention; definitions
of prespecified outcomes (of interest to this review); and the corresponding results (if applicable
confounder-adjusted estimates were given, with confounders for which was adjusted stated).
Items were extracted largely from the Strengthening the Reporting of Observational Studies in
Epidemiology (“STROBE”) statement8 and the Cochrane Handbook9. When there were questions
regarding these items, the authors of the articles in question were contacted for clarification.
Study quality and assessment of the strength of evidence. A quality assessment checklist was
constructed on the basis of the results of a systematic review evaluating tools for assessing quality
and susceptibility to bias in observational studies and the Cochrane Handbook regarding quality
assessment for randomized controlled trials9,10. Nine criteria were used across 5 quality domains.
The criteria for quality assessment can be found in Appendix Table 1. Studies were considered as
highly susceptible to bias if 2 or more of the 5 domains were scored as susceptible to bias, or if 3 or
more of the 5 domains were scored as unclear. The strength of the evidence for each intervention
4
was assessed by 2 reviewers (ST, SVV) according to predefined criteria adapted from the Canadian

Review of preconception lifestyle interventions

Task Force on the Periodic Health Examination11. In case of disagreement, a third reviewer (SD) was
asked to resolve the discrepancy. The applied classification for the strength of evidence can be
found in Appendix Table 1.

Data analysis
Because of presumed clinical heterogeneity, no attempt for a meta-analysis was prespecified.

RESULTS
Study identification and selection
From the search for articles related to preconceptional lifestyle interventions in women and
behavior change and/or risk factor modification and/or pregnancy outcome, 105 fulltext articles
were retrieved from 4,604 references (2,777 from Medline; 1,127 from Embase; 671 from Web of
Science; and 29 references from reviews). After exclusion of 61 full-text articles for stated reasons,
44 articles fulfilled the selection criteria (Figure 1).
Table 1 summarizes the included studies. Results are classified as follows. First, studies were
grouped by the core risk factor that the interventions address and report. Multiple risk factor
studies with multiple outcomes were classified separately. The rationale for this approach is to
give a structured overview and classification between studies addressing and reporting a single
risk factor versus multiple risk factor studies. Second, interventions were classified into individual
(individual consultation of a patient/couple), group-based (consultation of patients/couples
performed in groups), or collective interventions (interventions targeted at a group of people as
a whole, e.g., iodizing salt in prevention of hypothyroidism)12. The majority of studies focused on
individual interventions13–38, 3 studies focused on group interventions39–41, 1 study focused on a
mix of individual and group intervention42, and 14 studies focused on collective interventions43–56.

63
 FIGURE 1: Flowchart: identification, screening, and selection process of studies for inclusion in review.

4,575 Records identified through

Identification

29 records identified
database searching:
through citation tracking of
2,777 Medline
systematic reviews
1,127 Embase
671 Web of Science

1,267 Records after duplicates

4
removed
Screening

1,162 Records excluded after screening of titles /

abstracts

105 Records screened

Eligibility

105 Full text articles assessed for

1
eligibility Records excluded (when available based on full text):
intervention was not preconceptional n= 2
No intervention n= 48
No original data (review, editorial letter etc) n= 3
No outcome n= 5
Included

44 Studies included in qualitative Not available n= 3

synthesis

Of the 44 studies identified, 25 of those studies reported on pregnancy outcome13–23,30,34,44–54,56,

18 studies reported on behavior change regarding the risk behavior(s)24–29,32,33,35–43,55, and 1 study
reported on both pregnancy and behavior change outcome.31 Behavior change was most often
based on self-reported outcomes25–27,31,33,36,39,41–43; 1 study measured behavior change with
biomarkers only29, and 8 studies measured behavior change by using a combination of self-
report and biomarkers24,28,32,35,37,38,40,55; 19 randomized controlled trials13–22,25,26,30,31,36,38–41, 22 cohort
studies24,27–29,32,35,37,42–56, 1 case-control study34, 1 cohort controlled trial23, and 1 cross-sectional
study33 were identified. Results are presented in Appendix Table 2 and discussed per (risk) behavior
in the following section.

Alcohol
One randomized controlled trial reported on the effectiveness of a program to reduce alcohol-
exposed pregnancies by reducing risky drinking (8 drinks/week or >5 drinks on 1 occasion) in
women in whom conception could occur. Floyd et al.36 assessed the effectiveness of a prevention
program consisting of 4 counselling sessions with personalized feedback and goal setting
regarding risky drinking. Participants also received a counselling session on contraception. The
comparison group received written information regarding alcohol risks and women’s health. The
study population (n = 830) consisted of women of childbearing age not planning pregnancy who
were engaged in risky drinking. Women who received motivational counselling sessions and
counselling about contraception had significantly higher odds to be at reduced risk for an alcohol-

64
exposed pregnancy up to 9 months after intervention (odds ratio = 2.11, 95% confidence interval
(CI): 1.47,3 .03). This outcome is based on self-reported behavior change. Because the trial was
conducted in a population with a high predefined risk of alcohol consumption, results are limited
in generalizability. Regarding other criteria, the study quality was good resulting in a low risk of bias
overall. Considering the aims of the review, this study was not conducted specifically with women
planning a pregnancy. The strength of evidence is I-a.

Smoking
Three studies reported on the effectiveness of the advice to quit smoking: 1 randomized controlled
trial38 and 2 cohort studies29,35. One study assessed the effectiveness of a preconceptional health
program in terms of behaviour changes with a biomarker. Czeizel assessed smoking cessation
rates with urinary cotinine.29 The intervention, smoking cessation advice at a preconceptional
consultation, resulted in a decrease of smoking rates after 3 months (17.9% vs. 12.4%). In the
report by Hughes et al.38, the effectiveness of a “stage of change”– oriented, scripted hand out
4
and counselling at the hospital’s cessation clinic was assessed. The comparison group received

Review of preconception lifestyle interventions

information about the impact of prepregnancy smoking. De Weerd et al.35 evaluated provision of
advice to stop smoking. Both studies were performed in a hospital-based population; Hughes et al.
also included pregnant women. Outcomes were self-reported behavior change and were verified
with exhaled carbon monoxide measurements in the report by Hughes et al. and with the biomarker
cotinine in by de Weerd et al. With only advice to stop smoking, 88% of the self-reported smokers
reduced smoking; however, none ceased smoking. The stages of change-oriented counselling in
the report by Hughes et al. was not proven effective in the short term; however, after 12 months,
women in the intervention group were significantly more likely to maintain smoking cessation than
those in the control group. Although the selection of the study population was unclear in the study
by Czeizel29, overall susceptibility to bias was low. The study by de Weerd et al.35 is susceptible to
an attrition bias: how loss to follow-up is dealt with is unknown. As data collection was based on a
letter, items of quality assessment were unclear. Both Hughes et al.38 and de Weerd et al.35 sampled
patients within a hospital-based setting. The strength of evidence is I-a 38 and II-2. 29,35

Nutrition
Three studies focused on the effectiveness of a nutritional intervention program: 1 randomized
controlled trial39 and 2 case-control studies.34,42 Caan et al.34 assessed the effect of long-term
enrollment in the Women, Infants, and Children (WIC) supplemental food program with short-
term enrollment among women with low income and nutritionally at risk in their interpregnancy
interval. Long-term (5–7 months) WIC support was associated with a positive effect on birth weight
and birth length. Cena et al.39 assessed the effect of nutrition lessons regarding folate among
low-income, nonpregnant women. The comparison group underwent a lesson about resource
management. Nutrition lessons led to increased selfreported intake of dietary folate.
Doyle et al.42 assessed the effectiveness of a preconception nutrition counselling program
(educational group events, and nutrition newsletters) among women with pregnancy intention

65
 TABLE 1 Characteristics of studies reporting on the effectiveness of preconceptional interventions included in this systematic review (n=44; 1987-2012 Australia, Can-

ada, China, Denmark, Finland, France, Germany, Hungary, Israel, the Netherlands, Norway, Union of Soviet Socialist Republics, United Kingdom, United States)

Reported outcome
Study Study design Study population Behavioral change Pregnancy outcome High susceptibility to bias

I Alcohol
Individual Interventions Floyd 2007 35 RCT N= 593 Self reported No
II Smoking
Individual Interventions Hughes 2000 38 RCT N= 204 Self reported and biomarkers No
de Weerd 2001 35 Cohort N= 111 Self reported and biomarkers No Yes
III Nutrition
Individual interventions Caan 1987 34 CC N= 703 Not reported Yes
Hammiche 2011 37 Cohort N= 110 Self reported and biomarkers No
Group interventions Cena 2008 39 RCT N= 153 Self reported No

66
Mixed individual and
Doyle 1999 42 Cohort N= 41 Self reported No
group interventions
IV Folic acid
Individual interventions MRC Vitamin Study Re-
RCT N= 1,817 Not reported Yes Yes
folic acid advice and search Group, 1991 13
provision
Czeizel 1992 14 RCT N= 5,453 Not reported Yes Yes
Czeizel 1993 15 RCT N= 5,453 Not reported Yes Yes
Czeizel 1993 17 RCT N= 4,156 Not reported Yes
Czeizel 1994 20 RCT N= 5,453 Not reported Yes Yes
Czeizel 1996 16 RCT N= 5,453 Not reported Yes
Czeizel 1998 18 RCT N= 4,823 Not reported Yes Yes
Rolschau 1999 21 RCT N= 14,021 Not reported Yes
Ulrich 1999 22 RCT N= 8,184 Not reported Yes
Czeizel 2003 20 RCT N= 5,453 Not reported Yes Yes
Czeizel 2004 23 CCT N= 6,138 Not reported Yes
Watkins 2004 24 Cohort N= 165 Self reported and biomarkers No
 Robbins 2005 25 RCT N= 280 Self reported No
Schwarz 2008 26 RCT N= 265 Self reported No
Morgan 2009 27 Cohort N= 322 Self reported No
Individual Interventions
de Weerd 2002 28 Cohort N= 111 Self reported and biomarkers No
Folic acid advice

Collective interventions: Chan 2001 43 Cohort N= 512 Self reported No

public campaign Myers 2001 44 Cohort N= 222,314 Not reported Yes
Gindler 2001 45 Cohort N= 23,806 Not reported Yes
Collective interventions Gucciardi 2002 46 Cohort cases=3,207 Not reported Yes
fortification Honein 2001 47 Cohort N= 39,434,211 Not reported Yes
Persad 2002 48 Cohort N= 107,851 Not reported Yes
Ray 2002 49 Cohort N= 218, 977 Not reported Yes
Williams 2002 50 Cohort cases=5,630 Not reported Yes
CDC 2004 51 Cohort N= 6.9 million Not reported Yes
Liu 2004 55 Cohort N= 780 Self reported and biomarkers No
Canfield 2005 53 Cohort N= 9,729,763 Not reported Yes

67
Botto 2006 52 Cohort N= 1.5 million Not reported Yes
Yazdy 2007 56 Cohort N= 38,232 Not reported Yes
de Wals 2007 54 Cohort N= 1.9 million Not reported Yes
Programs
Individual Interventions Czeizel 1999 29 Cohort N= 8,837 Biomarkers No
Lumley 2006 30 RCT N= 786 Not reported Yes
Elsinga 2008 31 RCT N= 460 Self reported Yes
Ockhuijsen 2011 32 Cohort N= 101 Self reported No
Williams 2012 33 CSS N= 30,481 Self reported No
Group interventions Hillemeier 2008 40 RCT N= 360 Self reported and biomarkers No
N= 268 at 12 months
Weisman 2011 41 RCT Self reported No
N=262 at 6 months

Review of preconception lifestyle interventions

4
and history of a prior low birthweight baby and an inadequate dietary intake. The interventions led
to higher intake of certain micronutrients, except for folic acid from diet.
Doyle et al.42 sampled women in a hospital-based setting. Overall, the quality criteria were
assessed as good, resulting in a low risk of bias. The strength of evidence is II-2 34,42 and I-a.39

Folic acid
Thirty studies were identified as reporting on the effectiveness of folic acid supplementation
and fortification. Sixteen studies were individual-based programs, of which 12 were randomized
controlled trials13–22,25,26, 1 was a cohort controlled trial23, and 3 were cohort studies24,27,28; 14 cohort
studies43–56 were collective interventions, namely, folic acid fortification or public campaigns.
Regarding individual-based programs, 16 studies13–28 provided folic acid supplements. No studies
were restricted to advising only folic acid supplements.
From the 15 studies that provided supplements, there were 3 trials that provided folic acid
supplements as well as counselling on folic acid: A significant beneficial effect of selfreported folic
acid supplement use was shown.25–27
Counselling varied from brief folic acid counselling to a computerized educational session. Only
Morgan et al.27 succeeded in showing a significant increase in self-reported daily multivitamin intake.
Morgan et al. and Schwarz et al.26 showed an increase of self-reported use up to 6–10months after
the counselling and provision of supplements. Two trials, those by Watkins et al.24 and de Weerd et
al.28, assessed the effectiveness of folic acid supplement provision and counselling with biomarkers
in addition to selfreported outcomes. Watkins et al. did not show a significant increase in folic acid
use based on self-reported outcomes and serum folate levels before and after the intervention.
De Weerd et al. reported a significant increase of self-reported supplement use among women
planning a pregnancy. An elevated red cell folate level 4 months postintervention was found. All
trials were conducted among women of childbearing age; pregnancy intention was not always
specified. Susceptibility to bias was assessed as low; the strength of evidence is I-a 25,26 and II-2.24,27,28
Besides the effect of individual folic acid interventions on behavior change, 11 studies reported
on the effect on pregnancy outcome. No significant difference in miscarriage rates was shown in
1 study.13 Nine studies showed associations with a lower risk for certain congenital anomalies (e.g.,
urinary tract anomalies, cardiovascular anomalies, limb deficiencies, oral facial clefts, and urinary
tract defects, talipes, and hypospadias).14–20,22,23 One study reported a lower incidence of low birth
weight in the folic acid supplementation group; the trial did not show an effect on gestational birth
weight or preterm birth.21 Studies reporting on pregnancy outcomes varied in study quality. In
those by the Medical Research Council Vitamin Study Research Group13 and Czeizel et al.14–17,19,20,
many quality items were not clarified. Therefore, these studies were assessed as highly susceptible
to bias. The strength of evidence is I-a13–22 and II-2.23
Of 14 collective folic acid intervention studies, 3 cohort studies reported on the effectiveness of
a folic acid campaign.43–45 Chan et al.43 reported on behavior change, and Myers et al.44 and Gindler
et al.45 reported on pregnancy outcome.
Chan et al. investigated the effect of a folate campaign (information regarding the importance

68
of folic acid in the reduction of neural tube defects and pamphlets advertising available resources)
targeted to interconceptional women of reproductive age as well as health-care professionals.
Selfreported consumption of folate-rich food and folic acid tablet use periconceptionally increased
from 12% to 18.6% and from 10.1% to 26.7% 1 year after the campaign.
Myers et al.44 and Gindler et al.45 evaluated the effect of a public health campaign targeted
to women attending a premarital examination. The intervention in both studies included the
advice for women to take folic acid daily from the premarital examination until the end of the first
trimester of pregnancy. In Myers et al., supplementation was associated with a risk reduction of
41% in imperforate anus of the child. The study of Gindler et al. showed a higher relative risk of
miscarriages for women with folic acid use (relative risk = 1.03, 95%CI: 0.89, 1.20). Susceptibility to
bias was assessed as low; the strength of evidence is II-2.43–45
Regarding collective interventions, 11 large cohorts46–56 were included and reported on the
effectiveness of folic acid fortification. One study reported on behavior change assessed with
biomarkers55, and 10 studies evaluated changes in prevalence rates of congenital anomalies.46–54,56
4
Liu et al.55 evaluated the effectiveness of folic acid food fortification among women of

Review of preconception lifestyle interventions

childbearing age and seniors over 65 years, recruited prefortification through a random telephone
survey. Postfortification, the annual rate of neural tube defects was decreased by 78%, red blood
cell folate was significantly increased, and the proportion of women taking a vitamin supplement
containing folic acid was significantly increased from17% to 28%. Susceptibility to bias was
assessed as low; the strength of evidence is II-2.55
Eight large cohorts assessed the effectiveness of fortification in reduction of neural tube defect
prevalence rates46–52,54. Cases were retrospectively selected from birth certificate information and
registered databases. All studies showed a decline, ranging from 10% to 54%, in the incidence of
neural tube defects postfortification. Not all studies included stillborn and terminated pregnancies
in the time period assessed. Furthermore, subgroup analysis of spina bifida and anencephaly
showed a decrease in prevalence varying between 16% and 60%. Susceptibility to bias was
assessed as low; the strength of evidence is II-2 46–52,54.
Canfield et al.53 assessed prevalence rates of other congenital abnormalities besides neural tube
defects postfortification. A decrease in prevalence rates was noted for anencephaly, spina bifida,
transposition of the great arteries, cleft palate, pyloric stenosis, upper limb reduction defects,
omphalocele, and obstructive genitourinary defects. Susceptibility to bias was assessed as low; the
strength of evidence is II-2.53
Yazdy et al.56 showed a significant decline of 6% in orofacial clefts following folic acid
fortification in a subgroup of non-Hispanic whites. Susceptibility to bias was assessed as low; the
strength of evidence is II-2.56

Multiple risk factors

Seven studies were identified that reported on the effectiveness of multiple risk factors. The
majority of these studies were individual-based programs, of which 2 were randomized controlled
trials30,31, 2 were cohort studies32,37, and 1 was a cross-sectional study33; there were 2 group-based

69
randomized controlled trials.40,41
Williams et al.33 retrospectively assessed the effectiveness of receipt of PCC with regard to
preconceptional health behaviors. PCC was defined as any form of contact with a health-careworker
to prepare for a healthy pregnancy. The population consisted of interconceptional women planning
a pregnancy. Although the definition of PCC was broad, any receipt of PCC (content undefined)
led to a higher self-reported intake of multivitamins 1 month before pregnancy and cessation of
alcohol during the 3 months before pregnancy. Susceptibility to bias was assessed as low; the
strength of evidence is II-2.33
Hammiche et al.37 assessed the effectiveness of a tailored lifestyle and dietary consultation in
a hospital-sampled subfertile population that was planning pregnancy. Couples that attended a
second counselling session after 3 months reported a higher intake of fruit and fish and reduction
of their dietary risk score based on self-reported behaviors and biomarkers. However, only a
selection of the sampled subfertile patients within a hospital-based setting attended the second
consultation. Susceptibility of bias was assessed as low; the strength of evidence is II-2.
Ockhuijsen et al.32 assessed the effectiveness of PCC consultation in smoking cessation and
weight reduction among subfertile women. The outcome was self-reported smoking cessation and
self-reported weight reduction. With consultations every 4 weeks during a follow-up period varying
between 3 months and 1 year, 15 of 30 (50%) obese women lost weight (mean = 6.1 kg, standard
deviation, 3.6) and 7 of 23 (30%) women quit smoking. Because the follow-up period varied among
study participants, there is a susceptibility to a detection bias, as the study results are applicable
only to a hospital-based population of subfertile women. Overall susceptibility of bias was assessed
as low; the strength of evidence is II-2.
Two different multiple risk factor studies assessed effectiveness regarding pregnancy
outcomes.30,31 Lumley and Donohue30 assessed the effect of a home visit with prepregnancy
information, advice, and counselling given by midwives among low-income women in a community
setting. The intervention was compared with a postpartum home visit in which peripartum
experiences were discussed. Although birth weight was 97.4 g lower in the intervention group,
there was no significant difference in the outcomes: preterm birth (<32 weeks); low birth weight
(<2,500 g); and small for gestational age (birth weight, <10th percentile). Quantitative outcomes
showed a higher occurrence of preterm birth, low birth weight, and perinatal deaths. Because of
recruitment of women who were at high risk for poor birth outcomes, there is susceptibility for a
selection bias. Overall, the study was assessed as low susceptibility to bias; the strength of evidence
is I-a.
Elsinga et al.31 investigated the effectiveness of systematic PCC risk detection and intervention
compared with the standard care given by general practitioners among women contemplating
pregnancy. The study population consisted mainly of Dutch and high-educated women. The
outcome was self-reported behavior change and an adverse outcome of subsequent pregnancy.
Systematic counselling and intervention led to a significantly higher intake of folic acid and lower
alcohol consumption before pregnancy. Adverse pregnancy outcome (defined as premature birth
(<37 weeks), low birth weight (<2,500 g), small for gestational age (growth, <P2,3 (−2 standard

70
deviations)), and congenital anomalies) was 16.2% in the intervention group versus 20.2% in the
control group. The odds ratio for an adverse pregnancy outcome after preconception counselling
was 0.77 (95% CI: 0.48, 1.22). Susceptibility to bias was assessed as low; strength of evidence is I-a.
Hillemeier et al.40 assessed the effectiveness of a preconceptional group-based intervention
program regarding nutrition and physical activity among women “capable of becoming pregnant.”
The comparison group did not undergo any intervention. Women in the intervention group were
more likely to read food labels, to use a daily multivitamin that contains folic acid, and to meet
recommended levels of physical activity. However, half of the study population did not attend the
follow-up consultation and was excluded. Weisman et al.41 performed a follow-up study to assess
the maintenance of the aforementioned behavior changes 12-months after the intervention.
After 12 months, women in the intervention group were more likely to use a daily multivitamin
containing folic acid and to have a lower body mass index. Intervention effects on physical activity
were not maintained, and effects on reading food labels for nutritional values diminished between
the 6- and 12-month follow-up periods.
4
Allocation concealment in the studies by Hillemeier et al.40 and Weisman et al.41 was unclear,

Review of preconception lifestyle interventions

and patient sampling was unclear, resulting in a potential selection bias. Overall susceptibility to
bias was assessed as low; the strength of evidence is I-a.

Physical activity and weight loss

No studies reporting a specific intervention targeting physical activity and weight loss in a
preconceptional population were found. Numerous studies did find that, when preconceptional
health was addressed, this had a beneficial effect on physical activity in the short run37,40 and weight
loss.32

DISCUSSION
Regarding alcohol consumption, only 1 single risk factor study was available. Women who engaged
in risky behaviors reduced their alcohol consumption to less risky levels following a relatively
intensive intervention.36 However, a multiple risk factor approach in which reduction of alcohol
consumption was one of the targeted health behaviors in women contemplating pregnancy
was shown to be effective among highly educated women.31 The identified studies to assess the
effectiveness in altering behavior regarding alcohol consumption are proven effective for only
a selective group, and therefore more evidence is needed to justify that this intervention be
embedded in routine care. No studies reported on the effectiveness regarding pregnancy outcome.
The effectiveness of PCC in reducing preconceptional smoking cessation is not clear. Hughes
et al.38 reported only maintenance of smoking cessation, verified by a biomarker, in the long term
and only among a small subpopulation. A “stages of change” approach does not seem effective in
terms of cessation in the short term but could be considered to achieve maintenance of smoking
cessation. Results from other studies using a biomarker showed contradictory results: a decrease in
initial smokers29 versus no smoking cessation.35 However, a large proportion reduced smoking.35 No

71
studies reported on the effectiveness regarding pregnancy outcome.
Nutritional interventions seemed to be effective in changing dietary health behavior. However,
alterations were assessed only among a selective group of women, for example, women living
on food stamps with prior adverse pregnancy outcomes.39,42 Regarding the effect on pregnancy
outcome, longterm nutritional support was associated with a positive effect on birth weight.34 The
interventions provided in the available studies are proven effective only for a selective group, and
therefore more evidence is needed to justify that they be embedded in routine care for the general
public contemplating pregnancy.
Folic acid interventions were differentiated in individual advice and/or provision of folic acid
supplements and in collective interventions, such as public campaigns and food fortification
studies. Folic acid interventions proved to be effective in achieving self-reported intake of folic
acid when folic acid supplements were provided.25–27 The studies that assessed effectiveness of
provision of folic acid with serum folate as a biomarker were conflicting. However in one of these
studies, the self-reported outcomes did not support folic acid provision either.24 The other study did
show a slight increase in self-reported folic acid use and maintenance of levels biologically shown
by erythrocyte folic acid 4 months post intervention.28 On the basis of interventions provided in
the available studies, it remains unclear whether sole advice to take folic acid supplementation
is sufficient to achieve folic acid supplementation compared with the provision of folic acid
supplements. To compare the effectiveness of sole advice versus advice including provision of
folic acid supplements, a randomized controlled trial would need to be conducted, preferably
using biomarkers. Eleven randomized controlled trials reported on the effect of individual folic
acid advice, mostly including provision on pregnancy outcomes. On the basis of 1 study (with
nonsignificant outcomes), there does not appear to be an effect on the miscarriage rate.13 Nine
studies showed associations with a lower risk for certain congenital anomalies.14–20,22,23 One study
showed a lower incidence of low birth weight.21 Study quality items were unclear in a majority of
these studies; it is unclear to what extent results are applicable for the general public. Because folic
acid is widely proven to be effective in reduction of the risk for neural tube defects, this evidence
should be considered as further support for interventions to achieve folic acid supplementation
preconceptionally. The findings of the effectiveness of collective approaches regarding folic
acid are in line with findings from the individual interventions, described above, regarding the
behavior outcomes and effect on pregnancy outcomes. An increase of folic acid intake due to food
fortification was further supported by the finding that folate biomarkers increased among women
postfortification.55 Furthermore, a folate campaign was also effective in increasing self-reported
consumption of folate rich food and folic acid supplementation.43 Similar to the individual folic
acid studies, the campaigns and fortification studies showed reduced occurrence of congenital
anomalies, mainly neural tube defects.44,46–54,56
Studies on multiple risk factors in reducing risky health behaviors all seemed to be effective
in 1 or more targeted risk behaviors, for example, weight loss, reduction of the number of
cigarettes smoked, a higher daily consumption of fruit, fish, and multivitamins, and cessation of
drinking.31,32,37,40 However, the contents of the interventions were often not specified. One trial

72
succeeded in showing the effectiveness of a multiple risk factor approach on adverse pregnancy
outcomes among a higher educated group of women in the Netherlands.31 Further evidence is
necessary regarding the beneficial effects of a multiple risk factor approach for PCC above single
intervention studies.

Strengths and limitations

As the studies identified in the review contained clinically heterogeneous data, they therefore
could not be pooled. Clinical heterogeneity was a result of differences in interventions applied to
different (sub)populations in different settings. Tailoring interventions seems to be very important
in order to meet the demands of different populations; however, it does not allow meta-analysis
regarding this topic. The timeframe of the intervention within the pregnancy planning scheme was
often undefined or classified differently. The duration, method of follow-up, and reported outcomes
were reported differently by studies. For the aforementioned reasons, this review is descriptive in
nature.
4
The evidence from the studies included in this review is likely to be at some risk of bias.

Review of preconception lifestyle interventions

Although studies made efforts to reduce bias by aspects of study design such as accounting for loss
to follow-up and reporting predefined outcomes, the risk associated with unclear patient sampling
and unclear allocation concealment could not easily be addressed. Follow-up was insufficient
in a number of studies to measure change in health behavior. Missing data are a particular
problem in studies where women are followed over time, but they are mostly excluded from the
analysis. Possible outcomes become difficult to interpret and apply only for a subset of the study
population. Findings for those women who are followed up at all data collection points may not be
applicable to those women with missing data. For instance, missing data could reflect the fact that
an intervention was not feasible for all participants. As a result of missing data, overestimation of
the effect could be measured, because the subset of the study population is not representative of
the wider population. Ten25–27,31,33,36,39,41–43 of the 19 studies relied only on self-report for information
on behavior change postintervention. Self-reported outcomes may not be reliable.57 In this review,
the more recent studies introduced the use of biomarkers to assess behavior change. This seems a
very welcome introduction that should be integrated in further research on this topic. A drawback
with biomarkers is the diagnostic value regarding the degree to which behaviors have changed.
Furthermore, not all studies elaborated on the cutoff levels they applied in the interpretation of
biomarkers.29,38,40,55 As the majority of studies did not include follow-up of subsequent pregnancies
after the preconceptional intervention or maintenance of health behaviour change, the effect
on pregnancy outcome could often not be assessed. Because some studies were conducted in
specific populations (e.g., hospital based with subfertile women, women at higher risk for adverse
pregnancy outcomes, and low-income women), it is not clear how easy it would be to transfer
interventions to other settings or the general population.
Regarding adequate implementation of interventions, it should also be noted that many
studies do not describe the details of the intervention thoroughly. More information is necessary,
such as who delivered the intervention and how the intervention was exactly implemented to

73
ensure adequate implementation of interventions in the future.
Study populations often comprised women of childbearing age, without further elaboration
of pregnancy intention. It may be that the period with pregnancy intention is a window of
opportunity to change risky health behaviors.36 Women are potentially more motivated to change
their behaviour in order to have a healthy child. This motivation could be very crucial in the effect of
the intervention in achieving and maintaining behavior change.
Besides population and intervention characteristics, as stated above, organizational factors
in the setting are of great importance. These organizational factors are largely dependent on the
nation’s health-care infrastructure, insurance system, and socioeconomic factors.1 Articles in this
field often lack these details or lack reflection on the relation of these factors to the reported
findings. Transfer of this knowledge seems very valuable.

CONCLUSION
As evidence for preconceptional risk factors associated with adverse pregnancy outcomes is large,
there is a need for effective interventions to reduce these risk factors and improve pregnancy
outcomes. However, overall, on the basis of the available evidence, there is a relatively short
list of core interventions for which there is substantial evidence of effectiveness when applied
in the preconception period. Regarding alcohol, evidence is lacking for interventions in the
preconceptional period. Regarding nutrition, preconceptional interventions are effective in
terms of dietary change and birth weight. Smoking interventions are effective in achieving
smoking reduction in the preconception period. Regarding folic acid, individual interventions
and collective interventions to increase folic acid use are effective in terms of behavioral change
and improvement of pregnancy outcomes. The additional benefits of a programmatic approach
above a single intervention approach remain difficult to assess; there were no comparative studies.
Integration of single interventions into care is a challenging discussion for which implementation
studies are necessary.Naturally, despite the relatively short list of core interventions, health-care
providers should continue with information provision about the consequences and risks of risky
behaviour to couples wishing to conceive.
Recommendations for future research are as follows. Include 1) follow-up of pregnancy outcomes;
2) confirmation of self-reported outcomes, for instance, with biomarkers; 3) description of
determinants (such as contemplation of pregnancy) that are associated with effective or
ineffective treatment outcomes to supply information on the generalizability of findings; and 4)
provision of specific information regarding the content of interventions and the setting to guide
implementation of interventions.

CONFLICTS OF INTEREST
None declared.

74
ACKNOWLEDGEMENTS
We are grateful for the support of Louis Volkers for drafting the search strategy and conducting the
electronic search. We thank Megan Hempstead for the final revision of this review.

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Oct 2005;73(10):679-689.
54. De Wals P, Tairou F, Van Allen MI, et al. Reduction in neural-tube defects after folic acid fortification in Canada. N Engl J Med.
Jul 12 2007;357(2):135-142.
55. Liu S, West R, Randell E, et al. A comprehensive evaluation of food fortification with folic acid for the primary prevention of
neural tube defects. BMC Pregnancy Childbirth. Sep 27 2004;4(1):20.
56. Yazdy MM, Honein MA, Xing J. Reduction in orofacial clefts following folic acid fortification of the U.S. grain supply. Birth
Defects Res A Clin Mol Teratol. Jan 2007;79(1):16-23.
57. Sikkens JJ, van Eijsden M, Bonsel GJ, Cornel MC. Validation of self-reported folic acid use in a multiethnic population: results
of the Amsterdam Born Children and their Development study. Public Health Nutr. Nov 2011;14(11):2022-2028.

77
APPENDIX 1: Search Strategy

PubMed:
PubMed: (preconception*[tw] OR pre-conception*[tw] OR prepregnan*[tw] OR pre-pregnan*[tw] OR pregestation*[tw]
OR pre-gestation*[tw] OR periconception*[tw] OR peri-conception*[tw] OR interconception*[tw] OR inter-concep-
tion*[tw] OR interpregnan*[tw] OR inter-pregnan*[tw] OR intergestation*[tw] OR inter-gestation*[tw] OR internatal*[tw]
OR inter-natal*[tw]) AND (education*[tw] OR promotion*[tw] OR care[tw] OR cares[tw] OR caring*[tw] OR healthcar*[tw]
OR campaign*[tw] OR counsel*[tw] OR wellness*[tw] OR intervent*[tw]) AND (matern*[tw] OR mother*[tw] OR pater-
nal*[tw] OR father*[tw] OR parent*[tw] OR man[tw] OR men[tw] OR woman[tw] OR women[tw] OR couple*[tw]) AND
(eng[la] OR dut[la] OR ger[la] OR fre[la] OR spa[la]) NOT (animals[mesh] NOT humans[mesh])

Embase:
((preconception* OR prepregnan* OR pregestation* OR periconception* OR interconception* OR interpregnan* OR inter-
natal* OR intergestation* OR pre-conception OR pre-conceptional OR pre-pregnancy OR pre-pregnant OR pre-gestation
OR pre-gestational OR peri-conception OR peri-conceptional OR inter-conception OR inter-conceptional OR inter-preg-
nancy OR inter-pregnant OR inter-gestation OR inter-gestational OR inter-natal) NEXT/2 (education* OR promotion* OR
care OR cares OR caring* OR healthcar* OR campaign* OR counsel* OR wellness* OR intervent*)):ti,ab,de AND (matern*
OR mother* OR paternal* OR father* OR parent* OR man OR men OR woman OR women OR couple*):ti,ab,de AND ([eng-
lish]/lim OR [dutch]/lim OR [german]/lim OR [french]/lim OR [spanish]/lim) NOT ([animals]/lim NOT [humans]/lim)

Web of Science:
((preconception* OR prepregnan* OR pregestation* OR periconception* OR interconception* OR interpregnan* OR inter-
natal* OR intergestation* OR pre-conception OR pre-conceptional OR pre-pregnancy OR pre-pregnant OR pre-gestation
OR pre-gestational OR peri-conception OR peri-conceptional OR inter-conception OR inter-conceptional OR inter-preg-
nancy OR inter-pregnant OR inter-gestation OR inter-gestational OR inter-natal) NEAR/2 (education* OR promotion* OR
care OR cares OR caring* OR healthcar* OR campaign* OR counsel* OR wellness* OR intervent*)) AND (matern* OR moth-
er* OR paternal* OR father* OR parent* OR man OR men OR woman OR women OR couple*) NOT (animal* NOT [human*)
AND ([english]/lim OR [dutch]/lim OR [german]/lim OR [french]/lim OR [spanish]/lim)

78
APPENDIX TABLE 1: Quality Assessment Criteria and Assessment of Strength of Evidence

Quality assessment criteria: adapted from (9-10)

Domain Criteria
I Methods for selecting study par- The source population was appropriate AND in- or exclusion criteria were
ticipants defined.
II Methods for measuring expo- Methodology was adequate to detect stated outcomes
sure and outcome variables
III Design specific sources of bias a) randomized controlled trials: allocation was concealed
b) selection bias: patient selection and sampling (and in case of
a case-control or cross sectional study case selection or when
applicable matching) was adequate
c) detection bias: the length of follow-up was adequate to detect
outcome and equally applied amongst all groups
d) attrition bias: loss to follow-up/ drops outs were reported and 4
handled appropriately in analysis
e) reporting bias: outcomes were pre-specified and there was no

Review of preconception lifestyle interventions

selective report on outcomes
IV Statistical methods Statistical procedures were described adequately and if applicable adjust-
ment for confounding factors was reported
V Conflicts of interest Source of funding or conflicts of interests were reported
Classification of strength of evidence adapted from the Canadian Task Force for Preventive Medicine (11)
I-a: at least 1 properly conducted randomized controlled trial BEFORE pregnancy
I-b: at least 1 properly conducted randomized controlled trial not necessarily before pregnancy
II-1: well-designed controlled trials without randomization
II-2: cohort or case-control studies
II-3: multiple time series with or without intervention or dramatic results in uncontrolled experiments

79
 APPENDIX TABLE 2: Findings From Studies Reporting on the Effectiveness of Preconceptional Interventions per (Risk) Behavior (N=44)
Intervention
I Alcohol
Individual interventions
Floyd 200735 Aim: Assessment of efficacy of motivation-
al counselling to reduce the risk of an alco-
hol-exposed pregnancy.
Intervention: In the intervention group:
four motivational counselling sessions
(advice to reduce risky drinking, with per-
sonalised feedback and goal setting) and
a contraception counselling visit about
effective contraception use. The compari-
son group received written information on
alcohol and women’s health.
80
II SMOKING
Individual interventions
Hughes 200038 Aim: Assessment of effectiveness of a moti-
vational intervention to cease smoking and
maintain cessation.
Intervention: The intervention group re-
ceived scripted advice, “stage-of-change”
orientated hand-outs and a visit to a smok-
ing cessation clinic. The control group re-
ceived ‘standard care’ about the impact of
smoking.
De Weerd Aim: Assessment of efficacy of smoking
200134 cessation advice
Methods
Design: Randomized controlled trial.
Recruitment: Prospectively during 2002-
2004 in six community-based settings
(primary care, hospital clinics, and jails) by
media and presentations.
Follow-up: Assessment at baseline and at
three, six, and nine months following the
intervention by interview.
Design: Randomized controlled trial.
Recruitment: Prospectively from 1996 until
1999.
Data collection/ Follow-up: Questionnaire
and exhaled carbon monoxide measure-
ments at six and twelve months for both
groups.
Design: Cohort study.
Recruitment: Prospectively between 1997
and 1999. Women scheduled for an ap-
Population and setting
In- exclusion criteria: (1) Women aged 18-
44 years old capable of becoming preg-
nant; (2) sexual intercourse in the previous
three months without contraception with
a fertile man; (3) no condition causing
infertility; (4) not pregnant or planning
pregnancy at time of the intervention; (5)
engaged in risky drinking; and (6) available
for the follow-up period.
Study population: N= 830 (intervention
group n=416; comparison group n=414).
Loss to follow-up: N=125 in the interven-
tion group (63% attended all four counsel-
ling sessions); comparison group n=112.
These women were excluded in analysis.
Setting: Community-based setting: Flori-
da, Texas, and Virginia, USA.
Inclusion criteria: Newly referred infertile
and pregnant patients who had smoked
≥three cigarettes in the past six months;
not attending for genetic counselling or
habitual abortion.
Study population: N=94 infertile women
(intervention group n=47; comparison
group n=47) and n=110 pregnant wom-
en (intervention group n=56; comparison
group n=54)
Loss to follow-up: None.
Setting: Three university teaching hospitals
in Hamilton, Ontario, Canada.
In- exclusion criteria: Non-pregnant wom-
en scheduled for a preconception care ap-
pointment on the fertility clinic.
Strength of
Outcomes evidence
Outcomes: Odds for an alcohol exposed I-a
pregnancy (risky drinking: eight drinks/
week or >five drinks on one occasion and
ineffective contraception).
Results: Women in the intervention group
had higher odds to be at reduced risk for
an alcohol exposed pregnancy:
- At three months follow-up (OR 2.31; 95%
CI 1.69-3.20);
- At six months follow-up (OR 2.15; 95% CI
1.52-3.06); and
- At nine months follow-up (OR 2.11; 95%
CI 1.47-3.03).
Outcomes: Self-identified “stage-of- I-a
change” and rate of maintenance of cessa-
tion after twelve months.
Results: Intervention and comparison were
similarly effective; the rate of maintained
cessation rose significantly from 4% to 24%
over twelve months, with a mean delta
“stage-of-change” 0.28.
Outcomes: Smoking cessation or reduction II-2
by self-report and by cotinine levels.
Results: No self-reported smoking cessa

Intervention
Intervention:
Smoking cessation advice during a precon-
ceptional consultation three-four monthly.
III NUTRITION
Individual interventions
Caan 198734 Aim: Assessment of efficacy of the WIC pro-
gramme on birth outcomes.
Intervention: WIC support (food supple-
mentation and nutrition education) during
prior pregnancy and five to seven months
postpartum (intervention group) and up
to two months postpartum (comparison
81
group).
Hammiche 201137 Aim: Assessment of the efficacy of tailored
preconception counselling to modify die-
tary and lifestyle behaviors.
Intervention: A tailored dietary and life-
style consultation (focusing on folic acid
use, medication, alcohol, caffeine, drugs,
physical exercise, infection risk, body mass
index, waist circumference, waist to hip ra-
tio, blood pressure, vitamin B12, avoidance
of raw milk cheeses /raw meat or fish, and
rubella vaccination status).
Review of preconception lifestyle interventions
Methods
pointment were approached by letter for
inclusion. Patient sampling: consecutively.
Data collection/ Follow-up: Questionnaire
and blood sample at baseline and four-six
weeks later and every three-four months
until the sixth, eighth and twelfth week of
pregnancy.
Design: Case-control study.
Recruitment: Prospectively.
Data collection/ Follow-up: Until subse-
quent pregnancies up to 27 months after
the index birth were analysed.
Data was collected from medical and WIC
records.
Design: Cohort study.
Recruitment: Prospectively between 2007
and 2009.
Data collection/ Follow-up: Three months
after the consultation by questionnaires,
anthropometric measurements, and bi-
omarkers. Data was used to formulate a
personal Preconception Dietary Risk score
(PDR score) and a Rotterdam Reproductive
Risk Score (R3 score).
Population and setting
Study population: 16 self-reported smok-
ers and 24 smokers based on cotinine in
the serum ((cotinine >5µg/L) within a co-
hort of 111 women.
Baseline characteristics: unknown.
Loss to follow-up: Not known for the smok-
ing group. (Loss to follow-up in the initially
included population was N=33.)
Setting: Tertiary care setting, the Nether-
lands.
In- exclusion criteria: Women that had
received WIC support during their first
pregnancy and that gave birth to their first
infant after august 1981
Study population: N =897. Study and con-
trol groups were significantly different in
terms of race, underweight status, parity,
birth weight of the first child and birth in-
tervals. Correction was applied for these
confounders.
Loss to follow-up: N=178 were excluded
form analysis due to missing outcome
data, n=16 were excluded because of foe-
tal death or multiple birth.
Setting: 48/86 urban and rural Californian
local WIC agencies.
Inclusion criteria: Couples planning preg-
nancy that visited an outpatient academic
Obstetrics and Gynaecology clinic.
Study population: N=419 couples. Baseline
characteristics: median age 31 year; 56%
Dutch ethnicity; 35% high educated; and
93.8% of the couples were subfertile.
Loss to follow-up: 309 couples did not at-
tend the follow-up consultation.
Setting: Tertiary Obstetrics and Gynaecolo-
gy clinic in the Netherlands.
4
Strength of
Outcomes evidence
tion, however 88% of self-reported smok-
ers and 75% of cotinine validated smokers
reduced smoking.
Outcomes: The impact of five to seven II-2
months of WIC support compared to up
to two months of WIC support on birth
outcomes.
Results:
Five to seven months of WIC participation
was associated with a positive effect on:
- Birth weight (grams) adjusted for ges-
tational age (3461±26, 6 vs. 3341±27, 8;
p=0,003).
- Birth length (inch) (19,8±0,076 vs.
20,1±0,008; p=0,01).
Outcomes: Change in dietary and lifestyle II-2
behaviors and changes in the PDR- and R3-
score in women.
Results:
- Intake of fruit increased from 65 to 80%
in women.
- Recommended intake of fish increased
from 39 to 52% in women.
- Median PDR score decreased in women:
from 2.6 (95% CI 2.4-2.9) to 2.4 (95% CI
2.1-2.6)
 APPENDIX TABLE 2 (continued)
Intervention
Group interventions
Cena 200839 Aim: Evaluation of the effect of learn-
er-centered nutrition education on folate
intake and food-related behaviors.
Intervention: The intervention group un-
derwent a learner-centered nutrition les-
son with group discussions, participatory
activities, worksheets, visual aids, cooking
demonstrations, and instructor expla-
nations regarding recommended folate
intake and suppletion. The control group
received a lesson about resource manage-
82
ment.
Mixed individual and group interventions
Doyle 199942 Aim: Assessment of efficacy of a precon-
ception nutrition counselling programme.
Intervention: Nutritional assessment fol-
lowed by four to six weekly consultations
to strive to attain Reference Nutritional
Intakes (RNI) and additional monthly edu-
cational group events.
Methods
Design: Randomized controlled trial.
Recruitment: Prospectively starting in 2006
(duration unclear). Recruitment sites were
food stamp program offices; WIC clinics;
low-income schools; and community pro-
grams that serve low-income families or
individuals.
Data collection/ Follow-up: Data was col-
lected at baseline and four weeks after
each lesson, by questionnaire.
Design: Cohort study.
Recruitment: Prospectively from 1995 to
1996. Women were recruited during post-
partum hospital stay.
Data collection/ Follow-up: Seven-day die-
tary diary at baseline and six months later.
Population and setting
In- exclusion criteria: (1) Non-pregnant
women with a low-income (<185% of fed-
eral poverty level); (2) aged 18-45 years;
(3) English or Spanish understanding and
reading; and (4) primary purchaser of food
for herself or her family.
Study population: N=155 (intervention
group n=77; comparison group n=78).
Loss to follow-up: N=2. Group not speci-
fied.
Setting: Trained Food Stamp Nutrition Ed-
ucation staff in five Californian counties
(urban and rural).
In- exclusion criteria: Mothers who had
delivered a singleton baby weighing <2.5
kg (independent of gestational age), who
intended to have further pregnancies and
whose diets were assessed as being inad-
equate.
Study population: N=111.
Baseline characteristics: mean age 29.1;
ethnic origin African 27%, Asian 12%, Cau-
casian 41%, and West Indian 20%.
Loss to follow-up: 70 women did not fill
in the follow-up dietary diary, they were
excluded.
Setting: Dieticians and co-workers at a
Mother and Baby clinic.
Strength of
Outcomes evidence
- Median R3 score decreased in women
from 4.7 (95% CI 4.3-5.0) to 3.1 (95% CI 2.8-
3.4) due to less alcohol use (-14.6%), more
physical exercise and folic acid use.
Outcomes: Increases in folate intake (nat- I-a
ural food folate, synthetic folic acid from
fortified foods, synthetic folic acid from
supplements, total synthetic folic acid, and
total folate from all sources) and food relat-
ed behaviors compared to baseline.
Results:
- Significant increase in natural food folate
intake (p=0.009) and total folate from all
sources (p=0.045), compared to women in
the control group.
- Other results (synthetic folic acid from
fortified foods, synthetic folic acid from
supplements, and total synthetic folic acid)
were not significantly different.
Outcomes: Changes in dietary intake (Ref- II-2
erence Nutritional Intakes).
Results:
Post-intervention:
- A significant increase in the intake of pro-
tein, zinc, niacin equivalents and vitamin
B6.
- A small increase in the proportion of
mothers who met the dietary reference
values for energy and for 11 of the 15 nu-
trients considered (significance not tested).

Intervention
IV FOLIC ACID
Individual interventions: folic acid advice and provision
Medical Research Aim: To assess the efficacy of folic acid and/
Council Vitamin or multivitamin supplementation in the
Study Research prevention of neural tube defects.
13
Group 1991 Intervention: The following groups were
composed: (A) mineral+ folic acid, (B) min-
eral + folic acid + multivitamin, (C) mineral
+ placebo, (D) mineral + multivitamin (no
folic acid).
Czeizel 199214; Aim: To assess the efficacy of preconcep-
Czeizel 199315; tional multivitamin use in reduction of first
83
Czeizel 199417 and occurrence of NTD’s and other congenital
Czeizel 200320 defects.
Intervention: All women attended a Hun-
garian family planning pregnancy program
(a check up, consultation three months
later and consultation during pregnancy)
The intervention group received a box
with multivitamin tablets (0.8 mg folic acid,
twelve vitamins, four minerals and 3 trace
elements) with recommendation for 1
tablet a day preconceptional up to at least
the second missed menstrual period. The
control group: was recommended to take
a tablet with trace elements (manganese,
copper, zinc and a low dose of vitamin C)
daily.
Review of preconception lifestyle interventions
Methods
Design: Randomized controlled trial.
Recruitment: Prospectively between 1983
and 1991 from 33 participating centres.
Randomization: Method not specified.
Data collection/ follow-up: Clinical fol-
low-up was three monthly until the twelfth
week of pregnancy. At each visit number
of capsules taken was counted; blood and
urine samples were taken. Outcome of
pregnancies and health in first years of life
was assessed with annual questionnaires.
Design: Randomized controlled trial.
Recruitment: Prospectively, between 1984
and 1992. Women that visited the Hungari-
an family clinic were recruited.
Randomization: Patients were asked if they
agreed to their allocation on the basis of
the randomization table.
Data collection/ Follow-up: Clinical fol-
low-up was three monthly until pregnancy.
Self-reported supplement use, number
of tablets left was assessed. Pregnancy
outcomes were collected from certificates
filled in by women and verified by their
physician. . Follow-up regarding congenital
abnormalities not specified.
Population and setting
In-exclusion criteria: Women with a previ-
ous pregnancy affected by a neural tube
defects (not associated with the autosomal
recessive disorder or Meckel’s syndrome),
planning another pregnancy and not al-
ready taking vitamin supplements. Women
with epilepsy were excluded.
Study population: N=1,817 (group A
n=449, group B n=461, group C n=454,
group D n=453). Baseline characteristics:
no significant differences amongst groups.
Loss to follow-up: not reported.
Setting: Multicentre study in the United
Kingdom, Australia, Hungary, Israel, Cana-
da, France and USSR.
In-exclusion criteria: Women < 35years
planning pregnancy; not infertile, no previ-
ous wanted pregnancy.
Study population: N= 7,905 participants in
5,502 women a pregnancy was confirmed
(n=2,793 in the intervention group; n=
2,660 in the control group).
Baseline characteristics intervention group
vs. trace element group: age (mean years;
±SD) 27, ±3 vs. 27± 3; primiparous 88% vs.
89%; prepregnancy body weight 57.1kg
±7.7kg vs. 57.3 ±7.3 kg.
Loss to follow-up: 49/5,502 pregnancy out-
comes could not be clarified.
Setting: Hungarian Family planning clinics,
care delivered by qualified nurses in Hun-
gary.
4
Strength of
Outcomes evidence
Outcomes: Miscarriage rate. I-a
Results: No significant differences were
seen in miscarriage rates: RR=1.06; 95% CI
0.79-1.43, p=0.70 for groups A+B vs. C+D.
Outcomes: Pregnancy outcome: termina- I-A
tion rates, miscarriage rate (undefined), still
birth (> 28 weeks GA), live birth rate, low
birth weight rate (not defined), preterm
birth rate (not defined).
Results:
Birth outcomes:
- First trimester termination: n=6 (0.2%) vs.
n=6 (0.2%); second trimester termination
after diagnosis of fetal defect: n=3 (0.1%)
vs. n=13 (0.5%).
- Miscarriage: n=301 (10.8%) vs. n=251
(9.4%) (Χ2=2.69, p=0.10)
- Stillbirth n=11 (0.4%) vs. n=9 (0.3%).
- Live birth: n=2410 (86.3%) vs. n=2337
(n=87.9).
- Low birth weight rate: n-178 (7.5%) vs.
n=166 (7.2%) (Χ2=1.86, p=0.17).
- Preterm birth n=178 (7.5%) vs. n=166
(7.2%) (Χ2=0.21, p=0.64).
Congenital anomalies: in the article of
Czeizel, 1992; Prevalence’s of congenital
malformations intervention group (n=2104
 APPENDIX TABLE 2 (continued)
Intervention
84
Czeizel 199316 Aim: Assessment of effect of periconcep-
tional multivitamin supplementation on
neural tube defects (NTDs) and other con-
genital abnormalities.
Intervention: Intervention group: daily
multivitamin supplementation (containing
vitamins -including 0.8 mg of folic acid
– minerals and trace elements) precon-
ceptional until at least two months after
conception. The control group received a
daily trace element preconceptional until
pregnancy.
Methods
Design: Randomized controlled trial.
Recruitment: Prospectively. Women were
recruited via the Hungarian family plan-
ning programme.
Randomization: Method not reported.
Data collection/ Follow-up: until the end
of pregnancy (self-reported pregnancy
outcome. confirmed by their physician).
Infants were followed up with physical ex-
amination.
Population and setting
In-exclusion criteria: Women < 35years
planning pregnancy; not infertile, no previ-
ous wanted pregnancy.
Study population: N=4,753 confirmed
pregnancies (intervention group n=2,420;
comparison group n=2,333).
Baseline characteristics intervention group
vs. comparison group maternal age mean
26.8 [SD 3.4] vs. 26.7 [SD 3.3]; 94.5% vs.
94.0% primiparous women.
Loss to follow-up: 13% in the intervention
group and 12% in the comparison group
did not report pregnancy outcomes. The
analytic sample consisted of 4,156 women
(intervention group n=2,104; comparison
group n=2,052) and 3,713 infants (inter-
vention group n=1,876; comparison group
n=2,032).
Setting: Qualified nurses of the Hungarian
family planning programme.
Strength of
Outcomes evidence
in this subset) vs. comparison group
(n=2052 in this subset):
- Cardiovascular malformation: n=6 vs. n=9.
- Cleft lip and/or palate: n=4 vs. n=5.
- Hypospadias: n=1 vs. n=1.
- Obstructive defects of urinary system n=1
vs. n=2.
- Congenital postural deformity: n=2 vs.
n=0.
- Limb reduction defect: n=1 vs. n=5;
- Foramina parietale permagna: n=0 vs.
n=2.
- Exomphalos and gastroschisis: n=1 vs.
n=1.
- Hemangioma in the face: n=3 vs. n=1.
- Other: n=4 vs. n=7.
Outcomes: Number of major and mild con- I-a
genital abnormalities.
Results:
- The rate of major congenital abnormal-
ities other than NTDs and genetic syn-
dromes was 9.0/1000 in pregnancies with
known outcome in the vitamin group
and 16.6/1000 in the trace element group
(RR=1.85; 95% CI 1.02-3.38), difference
7.6/1000.
- The rate of all major congenital abnor-
malities other than NTDs and genetic syn-
dromes diagnosed up to the eight month
of life was 14.7/1000 informative pregnan-
cies in the vitamin group and 28.3/1000 in
the trace element group (RR=1.95; 95% CI
1.23-3.09), difference 13.6/1000.

Intervention
Czeizel 199618 Aim: Assessment of effect of periconcep-
tional multivitamin supplementation on
neural tube defects and other congenital
abnormalities.
Intervention: Intervention group: daily
multivitamin supplementation (containing
12 vitamins -including 0.8 mg of folic acid
– minerals and trace elements) precon-
ceptional until at least two months after
conception. The control group received a
daily trace element preconceptional until
pregnancy.
Czeizel 199819 Aim: To study the relation between per-
85
iconceptional use of multivitamins and
birth defects.
Intervention: All women attended a Hun-
garian family planning programme.
Intervention group: daily multivitamin sup-
plementation (containing vitamins -includ-
ing 0.8 mg of folic acid – minerals and trace
elements) preconceptional until at least
two months after conception. The control
group received a daily trace element pre-
conceptional until pregnancy.
Review of preconception lifestyle interventions
Methods
Design: Randomized controlled trial.
Recruitment: Prospectively. Women were
recruited via the Hungarian family plan-
ning programme from 1984 to 1992.
Randomization: unclear
Data collection/ Follow-up: until the end
of pregnancy (self-reported pregnancy
outcome. confirmed by their physician).
Infants were followed up with physical ex-
amination.
Design: Randomized controlled trial.
Recruitment: Prospectively between 1985
and 1993. Women were recruited via the
Hungarian family planning programme.
Randomization: method not reported.
Data collection/ Follow-up: until the end
of pregnancy (self-reported pregnancy
outcome. confirmed by their physician).
Infants were followed up with physical ex-
amination.
Population and setting
In-exclusion criteria: Women < 35 years
planning pregnancy; not infertile, no previ-
ous wanted pregnancy. Infants with minor
anomalies were excluded.
Study population: N=5,502 confirmed
pregnancies (intervention group n=2,819;
comparison group n=2,683). Baseline char-
acteristics intervention group vs. compar-
ison group: maternal age mean 26.9 [SD
3.4] vs. 26.9 [SD 3.3]; 88.3% vs. 89.9% prim-
iparous women.
Loss to follow-up: Dropout rate 0.9% in
both groups. The analytic sample consist-
ed of 5,453 women (intervention group
n=2,793; comparison group n=2,660) and
4,862 infants (intervention group n=2,471;
comparison group n=2,391).
Setting: Qualified nurses of the Hungarian
family planning programme.
In- exclusion criteria: women planning
pregnancy, not infertile.
Study population: N=4,862 (interven-
tion group n=2,471, comparison group
n=2,391). Baseline characteristics: not re-
ported.
Loss to follow-up: Intervention group 0.9%,
comparison group 0.9%.
Setting: Qualified nurses of the Hungarian
family planning programme.
4
Strength of
Outcomes evidence
Outcomes: Number of major congenital I-a
abnormalities.
Results:
- Major congenital abnormality: rate in the
intervention group: RR=0.51; 95% CI 0.36-
0.71, p<0.0001; after exclusion of neural
tube defects:
- After the exclusion of NTDs, the RR for a
major congenital abnormality in the in-
tervention group: 0.54; 95% CI 0.39-0.76,
p=0.0003.
Outcomes: Association of birth defects I-a
with maternal periconceptional multivita-
min use containing folic acid.
Results: Daily maternal periconceptional
multivitamin use was associated with:
- A non-significant risk reduction for orofa-
cial clefts: OR=0.77; 95% CI 0.22-2.69.
- A non-significant increased risk for cleft
lip with or without cleft palate: OR=1.29;
95% CI 0.32-5.22.
- A non-significant risk reduction for cleft
palate only: OR=0.19; 95% CI 0.01-4.03.
- A significant risk reduction for heart de-
fects: OR= 0.42; 95% CI 0.19-0.98.
- A non-significant risk reduction for out-
flow tract defects: OR= 0.48; 95% CI 0.04-
5.34.
- A significant risk reduction for conotrun-
cal defects: OR= 0.29; 95% CI 0.09-0.97.
- A non-significant risk reduction for limb
deficiencies: OR=0.19; 95% CI 0.03-1.18.
 APPENDIX TABLE 2 (continued)
Intervention
Rolschau 199921 Aim: To study the effect of two doses of fol-
ic acid suppletion on birth outcomes
Intervention: The intervention group re-
ceived free folic acid supplements (2,5mg)
for daily use; the control group received
free supplement (1.0mg) for daily use.
86
Ulrich 199922 Aim: To study prevalence of congenital ab-
normalities in a subset of Rolschau 1999.
Intervention: see Rolschau 1999.
Methods
Design: Randomized controlled trial.
Recruitment: Prospectively from 1983 to
1986. By campaign (mailings, media, and
involved health care professionals) ad-
dressing all women 18-35 years. Randomi-
zation: Not specified. Not all women were
randomized.
Data collection/ Follow-up: A registration
form was completed by the General prac-
titioner/ midwife/ doctor and kept by the
pregnant woman. Length of follow-up: up
to the infant’s medical examination at 5
months.
Design: subset of the study of Rolschau
1999, a randomized controlled trial.
Recruitment: See Rolschau 1999.
Randomization: Only randomized wom-
en in Rolschau 1999 were included in this
subset.
Population and setting
In-exclusion criteria: (1) Women aged 18-
35, (2) planning pregnancy or already preg-
nant (3) registered resident in the county
of Funen (4) that have not had a prior baby
with a NTD (5) multiples and abortions
were excluded.
Study population: N=8,184 women were
randomized; N=14,533 pregnancies in
8,184 women over the course of three
years, data was available regarding 14,021
pregnancies with 14,185 infants (13860
single pregnancies; 161 pluri pregnancies
with 325 infants). This study gives results of
13,860 single born infants.
Baseline characteristics: Total size of groups
and characteristics per group not present-
ed.
Loss to follow-up: Not accounted for.
Setting: Funen country, Denmark.
In-exclusion criteria: See Rolschau et al.
Study population: N=8,293 infants born
from 8,184 pregnancies. Baseline charac-
teristics: Folic acid use before pregnancy:
n=1,359 (16.6%) and during the first 19
weeks of pregnancy in n=6,825 pregnan
Strength of
Outcomes evidence
- A non-significant risk reduction for hy-
pertrophic pyloric stenosis: OR=0.24; 95%
CI 0.05-1.14.
- A significant risk reduction for urinary
tract defects: OR= 0.21; 95% CI 0.05-0.99.
- A significant risk reduction for obstructive
and renal agenesis: OR=0.12; 95% CI 0.02-
0.69.
Outcomes: Birth weight, incidence of pre- I-a
term, low birth weight (≤2,500 gram) and
small for gestational age (weight for gesta-
tional age <2SD than the average, exclud-
ing congenital anomalies and stillborn).
Results:
Intervention versus control group versus
no folic acid:
- Birth weight at gestational age: no sig-
nificant differences in gestational weight
(results not given); when FA was started
preconceptionally gestational birth weight
was higher.
- Preterm birth (excluding congenital ab-
normalities and perinatal deaths): no differ-
ence between different doses.
- Low birth weight: 40.9% vs. 51.5% and
59.8% p=0.01. There was no difference be-
tween users of 2.5mg and 1.0 mg FA.
- SGA: 14.7‰ when FA was started pre-
conceptionally and 17.4‰ when started
during the first 19 weeks of pregnancy. No
difference was observed between 2.5 and
1.0 mg FA.
Outcome: Selected congenital anomalies I-a
(neural tube defects (omitted from our re-
port) cardiovascular anomaly, limb reduc-
tion defects, urinary tract anomalies, oral
clefts, talipes, hypospadias).
Results: 2.5mg folic acid versus 1.0mg folic

Intervention
Czeizel 200423 Aim: To compare informative offspring of
women that used multivitamin supple-
ments and women that did not.
Intervention: The intervention group re-
ceived periconceptional suppletion with
daily multivitamin (containing 0.8 mg folic
acid) from at least one month before con-
87
ception. Supplements were provided. The
comparison group did not use suppletion.
Review of preconception lifestyle interventions
Methods
Data collection/ Follow-up: Complemen-
tary to the data collection for Part I of the
study (see Rolschau 1999) information on
congenital malformations was extracted
from (1) the European Registration of Con-
genital Anomalies and Twins (Eurocat) and,
(2) hospital records.
Design: Cohort controlled trial.
Recruitment: Prospectively from 1993 to
1996. Supplemented women were recruit-
ed from a Hungarian Preconceptional clin-
ic. Unsupplemented women were recruit-
ed at antenatal care clinics between the
8th and 12th week of pregnancy.
Matching: Unsupplemented women were
matched to supplemented women on base
of age, socioeconomic status, place of resi-
dence and year of pregnancy.
Data collection/ Follow-up: Questionnaires,
interview, personal notes, counting of re-
mainder of tablets. Pregnancy outcomes
were extracted from a pregnancy outcome
certificate. Congenital defects were detect-
ed in neonatal follow-up up to the twelfth
month and in autopsy reports of infant
deaths or stillborn infants.
Population and setting
cies (83.4%). There were n=224/8,293 in-
fants with congenital abnormalities.
Loss to follow-up: Not accounted for.
Setting: See Rolschau 1999.
Inclusion criteria: (1) Not sterile or sub-
fertile women; (2) capable of conception
within one year; (3) not currently pregnant
(4) women that didn’t use supplements
longer than 7 days were excluded.
Study population: N=6,246 pregnant wom-
en (n=3,123 supplemented and n=3,123
unsupplemented). Baseline characteristics:
not reported.
Loss to follow-up: Pregnancy outcomes
could not be clarified in 54 supplemented
and 47 unsupplemented women. There
were 3,069 pregnant women analysable in
each cohort.
Setting: 31 countryside centers in Hungary.
4
Strength of
Outcomes evidence
acid:
- Cardiovascular anomalies: 15/4165 ver-
sus 12/4128 versus. 13/2742.
- Limb reduction defects: 1/4165 versus
2/4128 versus 2/2742.
- Urinary tract anomalies: 9/4165 versus
4/4128 versus 2/2742.
- Oral clefts: 13/4165 versus 7/4128 versus
6/2742.
- Talipes: 3/4165 versus 6/4128 versus
6/2742.
- Hypospadias: 5/4165 versus 7/4128 ver-
sus 5/2742.
Outcomes: Occurrence of congenital ab- II-2
normalities (urinary tract anomalies, cardi-
ovascular anomalies, pyloric stenosis, limb
deficiencies, and oral facial clefts) amongst
folic acid supplemented and unsupple-
mented women in live born or still born
or terminated pregnancies in 2nd and 3rd
trimester.
Results:
Supplemented vs. unsupplemented:
- Cardiovascular malformations: n=31
vs. n=50 (OR=0.60; 95%CI 0.0.38-0.96);
Ventricular septum defect: n=5 vs. n=10
(OR=0.26; 95%CI 0.08-0.73); Conotruncal
defects (transposition of great vessels, Fal-
lot): n=3 vs. n=1 (OR=3, 95% CI 0.24-15.0);
Atrial septum defects Type II: n=9 vs. n=13
(OR=0.69; 95% CI 0.27-2.76).
- Urinary tract congenital anomalies: Renal
dysgenesis: n=2 vs. n=0; Cystic kidney: n=2
vs. n=0; Obstructive congenital anomalies:
n=2 vs. n=19 (OR=0.52; 95%CI 0.24-1.13).
- Congenital pyloric stenosis: n=0 vs. n=2.
- Limb deficiencies: n=1 vs. n=3 (OR=0.33;
95%CI 0.01-3.71).
- Orofacial clefts: n=4 vs. n=3 (OR=1.33;
95%CI 0.23 – 9.11).
 APPENDIX TABLE 2 (continued)
Intervention
Watkins 200424 Aim: To determine if folic acid consump-
tion is influenced by providing free folic
acid supplements or fortified breakfast
cereal to non-pregnant women of child-
bearing age.
Intervention: Provision of folic acid pills
(400 mcg), educational materials about fo-
lic acid, and super-fortified cereal (fortified
with 400 mcg of folic acid per serving).
Robbins 200525 Aim: To assess efficacy of a physician-based
intervention during routine gynaecologic
visits on women’s folic acid supplementa-
tion.
Intervention: The intervention group re-
88
ceived folic acid counselling and 30 folic
acid tablets from the gynaecologist at
start, a reminder phone call from a re-
search nurse 1 to 2 weeks after the coun-
selling and a pamphlet. The control group
received counselling on non-pregnancy
related preventive health behaviors; a
pamphlet on folic acid and a coupon for
free folic acid.
Schwarz 200826 Aim: To assess efficacy of a one-time
15-minute computer-assisted counselling
with provision of 200 free folate tablets in
increasing women’s use of folate supple-
ments.
Intervention: Computerized counselling
about periconception folate supplemen-
tation with nine questions and answers
in the form of short video segments. Each
subject was provided with 400 mcg folate
with written instructions to use daily. The
control group received counselling on
emergency contraception. Emergency
Methods
Design: Cohort study.
Recruitment: Prospectively between 2000
and 2001 from six family clinics in Georgia.
Patient sampling: unclear.
Data collection/ Follow-up: Brief survey
and blood sample after each clinic visit.
Design: Randomized controlled trial.
Recruitment: Prospectively. Women were
recruited via four clinics (two were aca-
demic and 2 were private practiced).
Randomization: At patient level – method
not described.
Data collection/ Follow-up: Self-reported
folic acid use at baseline and assessed by
phone interview 2 months later.
Design: Randomized controlled trial.
Recruitment: Prospective recruitment from
two urgent care clinics in San Francisco in
in 2005.
Randomization: Allocation occurred with
computer-generated sequences.
Data collection/ Follow-up: A phone survey
six months post enrolment.
Population and setting
In-exclusion criteria: Non-pregnant women
aged 18-45 years.
Study population: N=1,093.
Baseline characteristics: age 87.5% <35
years; race/ethnicity 54.1% White, 40.5%
Black, 4.9% Other; 48.9% high school grad-
uate or less; 32.4% smoker.
Loss to follow-up: N=928 visited only once;
response rate of 15.1%.
Setting: Six family planning clinics in Geor-
gia.
In-exclusion criteria: (1) Non-pregnant
women aged 18-45 years capable of be-
coming pregnant; (2) attending a routine
gynaecologic visit (not for preconception
care); (3) understanding English (4) women
with a pregnancy affected with a neural
tube defect in the past were excluded.
Study population: N=322 (intervention
group n=162; comparison group n=160).
Loss to follow-up: 13% (follow-up tele-
phone interview unsuccessful n=40 and re-
fused to complete the follow-up n=3). 279
patients completed the study (87%).
Setting: 2 academic and 2 private clinics.
In-exclusion criteria: English-speaking
women aged 18-45 years. Women unlikely
to become pregnant were excluded (she
was currently pregnant/ she had under-
gone a hysterectomy or tubal ligation/
intrauterine device in place/ vasectomized
partner) as were women without a phone
Study population: N=583 women were
screened; exclusion criteria applicable to
n=137; n=446 were randomized (inter-
vention group n=227, comparison group
n=219). Baseline characteristics: not speci-
fied, except that 4% stated that they were
Strength of
Outcomes evidence
Outcomes: Change in the self-reported use II-2
of folic acid and change in mean serum
folate levels.
Results: The interventions did not signif-
icantly influence self-reported folic acid
consumption (OR= 1.18; 95% CI 0.76-1.83)
or mean serum folate levels (results not
shown, p-values >0.1).
Outcomes: Changes in daily (7 days/week) I-a
and weekly (at least 1 day/week) folic acid
use.
Results:
- Weekly folic acid intake increased in the
intervention group by 68% vs. 20% in the
comparison group (p=0.008).
- No significant differences were found in
daily intake.
Outcomes: Folate supplements use In in- I-a
tervention group compared to the control
group.
Results: Folate supplement use in the in-
tervention group vs. the control group:
32% vs. 21%, RR=1.54; 95% CI=1.12-2.13;
p=0.007.
 Strength of
Intervention Methods Population and setting Outcomes evidence

contraception was provided with instruc- trying to become pregnant.

tions. Loss to follow-up: Intervention group
n=89, control group n= 92. Women were
less likely to complete the follow-up if
they were enrolled at the county clinic,
had lower incomes, had not completed a
college education, and were self-identified
as black.
Setting: Two semiprivate urgent care clinics
in San Francisco.

Morgan 200927 Aim: To determine if vitamin consumption
is influenced by providing a free bottle of
multivitamins to non-pregnant women of
childbearing age.
Intervention: Verbal counselling, provision
of written materials, and free bottles of
multivitamins containing 400 mcg folic
acid.
Design: Cohort study.
Recruitment: Prospectively in 2004 from 24
participating health departments. Patient
sampling: a proportional-to-size sampling
design gathered 3,500 consent forms of
which 14% were randomly chosen.
Data collection/ Follow-up: Eight to ten
months after the intervention a phone call
for an eight-question survey and some-
times a written survey was conducted.
In-exclusion criteria: Non-pregnant females
of childbearing age were included.
Study population: N=500. Baseline charac-
teristics: ethnic origin 60.3% White, 26.6%
Latino/Hispanic, 7.5% African American,
2.8% American Indian and 3.4% Other/un-
known; age 51.6% <25 years, 34.8% 25-34
years, 13.6% >34 years.
Loss to follow-up: N=178 did not complet-
ed the survey and were excluded of the
Outcomes: Change in the self-reported use II-2
of multivitamins at eight to ten months
follow-up.
Results:
A greater than two-fold increase (PR=2.1;
p<0.001) of daily multivitamin use (25%
vs. 53%).

89
analysis (response rate of 64.4%).
Setting: Nurses provided care in 24 local
county health departments in Western
North Carolina.

Individual Interventions: folic acid advice

de Weerd 200228 Aim: To assess whether counselling of
women planning pregnancy to start or
continue folic acid supplementation im-
proves folate status.
Intervention: Preconceptional advice to
take multivitamin containing 400µg of fo-
lic acid daily as part of a preconceptional
health program containing health promo-
tion (smoking cessation, nutritional habits,
information on the antenatal care system).
Design: Cohort study.
Recruitment: Prospectively in 1997-1999.
Couples were recruited from the fertility
clinic or clinic for preconception care clinic.
Data collection/ Follow-up: (1) A question-
naire at baseline (2) blood samples before
the first consultation and every three - four
months after counselling and in the sixth,
eighth and twelfth week of pregnancy.
Inclusion criteria: Women planning preg-
nancy and visiting preconception care
clinic (because of previous obstetric com-
plications or other maternal risk factors) or
fertility clinic.
Study population: N=111.
Baseline characteristics: mean age (±SD)
32.5 ±3.5 years; nulliparous 59.5%; White
race 97.3%; education level low 13.3%,
middle 40%, high 23.8%.
Loss to follow-up: Eligible women n=168.
Withdrawal: n=23 (reasons not stated),
n=33 losses to follow-up (unspecified), n=1
participant was excluded (missing data).
Setting: University hospital setting in the
Outcomes: Changes in self-reported folic II-2
acid supplementation (any frequency) and
red cell folate level (user status was defined
as ≥590 nmol/L) and serum folate (user sta-
tus defined as levels ≥20 nmol/L).
Results:
- Self-reported supplement use four
months after counselling: 54% (compared
to 56% of the women at baseline).
- Red cell folate level amongst non-users
four months after counselling: 680 nmol/L
(SD 52 nmol/L) (compared to 540 nmol/L
SD± 30 nmol/L). P<0.01.
- After one year no significant changes
were seen in folate status compared to

Review of preconception lifestyle interventions

4
 APPENDIX TABLE 2 (continued)
Intervention
Collective interventions: public campaign
Chan 200143 Aim: To study the effect of a folate cam-
paign towards targeted health profession-
als and women of reproductive age.
Intervention: Key messages of the folate
campaign were:
1) folate/ folic acid intake by women of
reproductive age reduces NTD’s in the off-
spring;
2) green leafy vegetables, fruit, cereals con-
tain the most folate;
3) folate/ folic acid intake is important in
the periconceptional phase.
90
These messages were incorporated into
information leaflets for professionals, con-
sumer pamphlets, guidelines, newsletter,
study curricula, presentations towards
professionals.
Myers 200144 Aim: To evaluate the effect of public health
campaign with periconceptional maternal
daily consumption of 400 mcg of folic acid
and the risk for imperforate anus in the
offspring.
Intervention: Intake of daily one pill con-
taining 400 mcg of folic acid from the time
of premarital examination of women and
until the end of the first trimester of preg-
nancy.
Methods
Design: Cohort study.
Recruitment: Retrospectively from 1994-
1998. Recruitment of two groups: (1) wom-
en through random dialling and (2) wom-
en post-delivery consecutively.
Data collection/ follow-up: Folic acid intake
by computer assisted telephone interviews
pre-campaign (1994) and during the cam-
paign (1995, 1996 and 1998) and folate use
with postnatal questionnaires (delivery:
1996-1998).
Design: Cohort study.
Recruitment: Prospectively between 1993
and 1995. Women were recruited from the
pregnancy monitoring system.
Follow-up/ data collection: Free bottles of
folic acid to women; three blinded pedia-
tricians reviewed birth reports of liveborn
and stillborn infants of at least 20 weeks’
gestation.
Population and setting
Netherlands. Counselling was performed
by a physician.
Inclusion criteria: Women aged 15-44, post-
natal, that delivered in 1995 and 1996.
Study population: N=512.
Baseline characteristics not provided; char-
acteristics are reported to be the same in
all pre- and post-campaign measurements.
Baseline characteristics: not reported.
Loss to follow-up: Not applicable.
Setting: Health professionals approached
for the campaign were general practi-
tioners, pharmacists, dieticians, nurse and
medical staff of the child and youth health
service in South Australia.
In-exclusion criteria: All women who regis-
tered with the pregnancy monitoring sys-
tem were included. Multiple pregnancies
were excluded.
Study population: N=222,314 (n=126,783
women taking periconceptional folic acid;
n=95,531 not taking periconceptional fo-
lic acid). Baseline characteristics North vs.
South: mean age 25.2 years vs. 25 years.
Loss to follow-up: Not applicable.
Setting: Village health care workers in two
provinces of China.
Strength of
Outcomes evidence
initial precounselling folate levels: women
that already used supplementation pre-
counselling maintained their folate use.
Outcomes: Folate consumed before and in II-2
the first three months of pregnancy.
Results:
- Women reported to have increased folate
rich food consumption: in 1995 12.0% vs.
18.6% in 1996.
- Self-reported periconceptionally folic acid
use of women increased: 1995 10.1%, 1996
26.7%, 1998 46.1%.
Outcomes: Association of imperforate anus II-2
with maternal periconceptional folic acid
supplementation.
Results:
Periconceptional daily 400 mcg folic acid
supplementation was, after controlling for
maternal age, associated with a risk reduc-
tion of 41% in imperforate anus of the child
(OR=0.59; 95% CI 0.33-1.07).

Intervention
Gindler 200145 Aim: To evaluate the effect of public health
campaign with periconceptional maternal
daily consumption of 400 mcg of folic acid
and the occurrence of miscarriages.
Intervention: Intake of daily one pill con-
taining 400 mcg of folic acid from the time
of premarital examination of women and
until the end of the first trimester of preg-
nancy.
Collective interventions: fortification
Gucciardi 200246 Aim: To study trends in the total incidence
of open neural tube defects in Canada cap-
91
turing the fortification period.
Intervention: Folic acid fortification of grain
is mandatory as of 1998.
Review of preconception lifestyle interventions
Methods
Design: Cohort study.
Recruitment: Prospectively between 1993
and 1995 from the pregnancy monitoring
system.
Follow-up/ data collection: Free bottles of
folic acid to women; booklet with data on
past pregnancy history, the prenatal period
and delivery.
Design: Cohort study.
Recruitment: Population based retrospec-
tively from 1986 to 1999 from the Canadian
Congenital Anomalies Surveillance System
and hospital data.
Data collection/ follow-up: Collection of
the total incidence of NTDs by combin-
ing the numbers of NTDs occurring in live
births, stillbirths and therapeutic abor-
tions.
Population and setting
Inclusion criteria: (1) Women residing in
Jiaxing City who had a premarital exami-
nation, (2) primigravid (3) who registered
with the pregnancy monitoring system.
Study population: N=23,806 (n=21,935
women taking periconceptional folic acid;
n=1,871 not taking periconceptional folic
acid). Baseline characteristics periconcep-
tional folic acid use vs. no use of folic acid:
age at pregnancy 23.5 years SD 1.5 vs. 23.8
years SD 2.1; BMI mean 20.4 SD 2.2 vs. 20.5
SD 2.2; high school education 5.3% vs.
11.4%; farmer 55.6% vs. 54.8%.
Loss to follow-up: Not applicable.
Setting: Village health care workers in Chi-
na.
Inclusion criteria: All live births, stillbirths
and therapeutic abortions with neural
tube defects registered in the databases
and hospital data. Therapeutic abortions
in free-standing abortion clinics were not
included.
Study population: Total neural tube defects
n=3,207 (live births n=1,503; stillbirths
n=425; therapeutic abortions n=1,279).
Baseline characteristics: not reported.
Loss to follow-up: Not applicable.
Setting: Ontario, Canada.
4
Strength of
Outcomes evidence
Outcomes: Association of miscarriages II-2
with maternal periconceptional folic acid
use.
Results:
Compared with women who had no folic
acid use, the RR of miscarriage for women
with periconceptional folic acid use was
1.03; 95% CI 0.89-1.20.
Outcomes: Changes in neural tube defects II-2
birth incidence per 10,000 (live births and
stillbirths only).
Results:
- Total neural tube defect incidence rate
increased by 38% from 1986 to 1995 (11.7
to 16.2/10000 pregnancies, p<0.001);
decreased by 47% from 1995 to 1999
(8.6/10000 pregnancies, p<0.001). – neural
tube defect birth incidence decreased by
50% from 1986 to 1999 (10.6 to 5.3/10000
births, p<0.001).
- Rate of neural tube defects in live births
declined by 50% from 8.6/10000 live births
in 1986 to 4.3/10000 live births in 1999
(p<0.001).
- The rate of stillbirths with a neural tube
defect decreased by 53%, from 33.6/1000
stillbirths in 1986 to 15.9/1000 stillbirths in
1999 (p<0.001).
- From 1986 to 1995 the rate of therapeutic
abortions in which a neural tube defect or
hydrocephalus was detected rose 190%,
 APPENDIX TABLE 2 (continued)
Intervention
Honein 200147 Aim: To study the effect of folate food for-
tification with folic acid on neural tube de-
fect birth prevalence.
Intervention: Folic acid fortification of grain
is mandatory as of 1998. The comparison
group was born prior to the fortification
period.
92
Persad 200248 Aim: To assess changes in annual incidence
of open neural tube defects, in Nova Scotia
after the introduction of folic acid fortifica-
tion.
Intervention: Folic acid fortification of grain
is mandatory as of 1998.
Methods
Design: Cohort study.
Recruitment: Population based retrospec-
tively between 1990 and 1999.
Patient sampling: pre-fortification group
(infants born from October 1995-1996) vs.
post-fortification group (infants born from
October 1998-1999).
Data collection/ Follow-up: Cases with in-
fants with spina bifida and anencephaly
were collected from birth certificate infor-
mation.
Design: Cohort study.
Recruitment: Population based retro-
spectively from 1991 to 2000 from the
Nova Scotia Atlee Perinatal Database and
from the Fetal Anomaly Database. Patient
sampling: pre-fortification selected all
open neural tube defects registered in
1991-1997 vs. post-fortification selected
all open neural tube defects registered in
1998-2000.
Data collection/ Follow-up: Collection of
annual incidence rates pre- and post-for-
tification.
Population and setting
In- exclusion criteria: (1) Infants with spi-
na bifida/ anencephaly or any congenital
abnormality, (2) born in 45 states (that
registered congenital anomalies on the
certificate).
Study population: N= 39,434,211. Baseline
characteristics: not provided.
Loss to follow-up: Not applicable.
Setting: 45 states of the U.S.A.
In-exclusion criteria: All live births, still-
births and terminated pregnancies with
open neural tube defects over a 10-year
period registered in the databases.
Study population: Total births n=107,851;
cases n=239.
Baseline characteristics: not reported.
Loss to follow-up: Not applicable.
Setting: Nova Scotia, Canada.
Strength of
Outcomes evidence
from 17.5 to 50.7 per 10000 abortions
(p<0.001). By 1999 it had fallen by 43% to
28.7/10000 abortions (p<0.001). This re-
sulted in a decrease in the ratio of neural
tube defect-affected births to therapeutic
abortions from 3:1 in 1986 to 1:1 in 1999.
Outcomes: Comparison of pre- and II-2
post-fortification spina bifida and anen-
cephaly prevalence rates.
Results: Decline in total neural tube defects
post-fortification compared to pre-fortifi-
cation: 19% (PR= 0.81; 95 CI 0.75-0.87).
Outcomes: Annual incidence changes of II-2
open neural tube defects, and of the sub-
groups spina bifida and anencephaly, per
1000 births (live births, stillbirths and ter-
minations) pre- and post-fortification.
Results:
- The total annual incidence of open neural
tube defects fell by 54% after folic acid for-
tification, from 2.58/1000 births on average
during 1991-1997 to 1.17/1000 births dur-
ing 1998-2000: RR=0.46; 95% CI 0.32-0.66;
p<0.001.
- Subgroup analysis of spina bifida, the
mean annual incidence decreased from
1.51/1000 births before to 0.62/1000 births
after fortification: RR=0.40; 95% CI 0.25-
0.67; p<0.001.
- Subgroup analysis of anencephaly, the
mean annual incidence decreased from
0.93/1000 births to 0.38/1000 births after
fortification: RR=0.41; 95% CI 0.22-0.77;
p=0.0004.

Intervention
Ray 200249 Aim: To assess the effect of folic acid food
fortification before and after on the preva-
lence of open neural tube defects.
Intervention: Folic acid fortification of grain
is mandatory as of 1998.
Williams 200250 Aim: To assess the effect of folic acid food
93
fortification before and after on the preva-
lence of spina bifida and anencephaly.
Intervention: Folic acid fortification of grain
is mandatory as of 1998.
CDC 200451 Aim: To assess the effect of folic acid food
fortification before and after on the preva-
lence of neural tube defects.
Intervention: Folic acid fortification of grain
Review of preconception lifestyle interventions
Methods
Design: Cohort study.
Recruitment: Population based retrospec-
tive from 1994 to 2000 from the Ontario
MSS database. Patient sampling: pre-for-
tification selected neural tube defects
registered 1994-1997 vs. post-fortification
selected neural tube defects registered
1998-2000.
Data collection/ follow-up: Antenatal di-
agnosed open neural tube defects (based
on ultrasonography or fetal autopsy after
therapeutic termination) and postnatal di-
agnosed open neural tube defects (based
on all live born and stillborn affected in-
fants after 20 weeks’ gestation) pre- and
post-fortification.
Design: Cohort study.
Recruitment: Population based retro-
spective from 1995 to 1999 from 24 birth
defects population-based surveillance
systems. Patient sampling: pre-fortification
1995-1996 or during optional fortification
1997-1998 or during mandatory fortifica-
tion 1998-1999 registered selected neural
tube defects.
Data collection/ follow-up: Collection of
the prevalence of neural tube defects (live
births, stillbirths, fetal deaths, and elective
terminations) using data from nine surveil-
lance systems with prenatal ascertainment
and data from 13 surveillance systems
without prenatal ascertainment and elec-
tively terminated cases during the three
periods.
Design: Cohort study.
Recruitment: Population based retrospec-
tive from 1995 to 2000 from 23 popula-
tion-based surveillance systems. Patient
Population and setting
In-exclusion criteria: All antenatal and
postnatal diagnosed open neural tube
defects among women residing in Ontario
and registered in the MSS database were
included.
Study population: N=218,977 and
n=117,986 women were screened for open
neural tube defects pre- and after fortifi-
cation respectively. Cases of open neural
tube defects n=248 and n=69 pre- and
after fortification respectively. Baseline
characteristics pre- vs. after fortification:
maternal age mean 30.1 years (SD 0.16) vs.
30.3 years (SD 0.081).
Loss to follow-up: Not applicable.
Setting: Ontario, Canada.
In-exclusion criteria: All antenatal and
postnatal diagnosed open neural tube
defects among women residing in Ontario
and registered in the MSS database were
included.
Study population: N=5,630 cases of spina
bifida and anencephaly from 1995-1999.
Baseline characteristics: not reported.
Loss to follow-up: Not applicable.
Setting: 24 states in the United states of
America.
In-exclusion criteria: All antenatal and
postnatal diagnosed neural tube defects
among women residing in Ontario and
registered in the MSS database were in
4
Strength of
Outcomes evidence
Outcomes: Decrease in the prevalence of II-2
open neural tube defects per 1000 preg-
nancies after fortification.
Results:
- The prevalence of open neural tube de-
fects decreased from 1.13/1000 (n=248)
pregnancies before fortification to
0.58/1000 (n=69) pregnancies thereafter:
PR=0.52; 95% CI 0.40-0.67, p<0.0001.
- The prevalence of anencephaly decreased
from 0.38/1000 (n=84) pregnancies before
fortification to 0.16/1000 (n=19) pregnan-
cies thereafter: PR=0.42; 95% CI 0.26-0.69,
p<0.0002.
- The prevalence of spina bifida decreased
from 0.75/1000 (n=164) pregnancies be-
fore fortification to 0.42/1000 (n=50) preg-
nancies thereafter: PR=0.57; 95% CI 0.41-
0.78, p<0.0001.
Outcomes: Decrease in the prevalence of II-2
neural tube defects after fortification.
Results:
Prevalences from the pre- to the mandato-
ry fortification period:
- Spina bifida decreased 31%: PR=0.69;
95% CI 0.63-0.74.
- Anencephaly decreased 16%: PR=0.84;
95% CI 0.75-0.95.
- Spina bifida decreased 40%: PR=0.60; 95%
CI 0.51-0.71 among the nine programs with
prenatal ascertainment and 28%: PR=0.72;
95% CI 0.65-0.80 among the thirteen pro-
grams without prenatal ascertainment.
Outcomes: Decrease in the prevalence of II-2
neural tube defect affected pregnancies
after fortification.
Results:
 APPENDIX TABLE 2 (continued)
Intervention
is mandatory as of 1998.
94
Liu 200455 Aim: To evaluate the effectiveness of the
public health strategy of food fortification
with folic acid and to determine possible
adverse effects resulting from fortification.
Intervention: Folic acid fortification of
white flour, pasta, and cornmeal.
Methods
sampling: pre-fortification selected neu-
ral tube defects registered 1995-1996 vs.
post-fortification selected neural tube de-
fects registered 1999-2000.
Data collection/ follow-up: Collection of
the prevalence of neural tube defect-af-
fected pregnancies (live births, stillbirths,
fetal deaths, and elective terminations)
using data from eight surveillance systems
with prenatal ascertainment and data
from fifteen surveillance systems without
prenatal ascertainment pre- and post-for-
tification.
Design: Cohort study.
Recruitment: Prospectively between 1997
and 1998. Women and seniors were recruit-
ed through a random telephone survey
prior to folic acid fortification.
Follow-up/ data collection: Women and
seniors were followed-up to two years
after the start of fortification In person in-
terviews recollected vitamin supplement
use, a Willet food frequency dietary ques-
tionnaire was taken to assess intake over
the past year, and blood samples pre- and
Population and setting
cluded.
Study population: N=6.9 million pregnan-
cies in 1999. Baseline characteristics: not
reported.
Loss to follow-up: Not applicable.
Setting: United States of America.
In-exclusion criteria:
- Women: non-pregnant of childbearing
age (19-44 years) not taking folic acid sup-
plements were included.
- Seniors: aged 65 years and over without a
vitamin B12 deficiency or anaemia, and not
taking vitamin B12 or supplements con-
taining folic acid were included.
Study population: Pre- fortification n=233
women, n=202 seniors; post-fortification
n=204 women, n=186 seniors. Baseline
characteristics: not reported.
Strength of
Outcomes evidence
- Estimated number of neural tube de-
fect-affected pregnancies declined from
4000 in 1995-1996 to 3000 in 1999-2000.
Data from systems with prenatal ascertain-
ment showed:
- Before fortification the total annual num-
ber of neural tube defect-affected preg-
nancies was 4130: n=2490 spina bifida and
n=1640 anencephaly.
- After fortification the total annual num-
ber of neural tube defect affected preg-
nancies was 3020: n=1640 spina bifida and
n=1380 anencephaly.
–> 27% decline.
Data from systems without prenatal ascer-
tainment showed:
- Before fortification the total annual num-
ber of neural tube defect-affected live
births, stillbirths, and fetal deaths at >20
weeks’ gestation was 2950: n=1980 spina
bifida and n=970 anencephaly.
- After fortification the total annual num-
ber of neural tube defect -affected live
births, stillbirths, and fetal deaths at >20
weeks’ gestation was 2180: n=1340 spina
bifida and n=840 anencephaly.
–> 26% decline.
Outcomes: Neural tube defect prevalence, II-2
red blood cell folate and dietary folate in-
take pre vs. post fortification.
Results:
- The total annual rate of neural tube
defects fell by 78% after the implemen-
tation of folic acid fortification (pre for-
tification: 4.36/1000 births; post fortifi-
cation 0.96/1000 births; RR=0.22; 95% CI
0.14-0.35; p<0.0001). Neural tube defects
amongst terminated pregnancies were sta-
ble throughout the pre- and post-forti
 Strength of
Intervention Methods Population and setting Outcomes evidence

post-fortification were taken.
Loss to follow-up: Not accounted for (wom-
en n=29; seniors n= 16).
Setting: Rural and urban sites of New-
foundland, Canada.
fication period.
- Significant increases in RBC folate levels
for women and seniors after mandatory
fortification (625 mol/L [SD 601-649] vs.
818 [SD 784-854]; p<0.001).
- The proportion of women aged 19-44
years taking a vitamin supplement con-
taining folic acid increased from 17% to
28% (p<0.003).

Canfield 200553 Aim: To assess changes in birth defect prev-
alences since folic acid fortification.
Intervention: Folic acid food fortification
after 1997.
Design: Cohort study.
Recruitment: Population based retrospec-
tive between 1995 and 2000 from popu-
lation-based registries. Patient sampling:
pre-fortification selected birth defects in
1995-1996 vs. post-fortification selected
birth defects in 1999-2000.
Data collection/ follow-up: Collection
of prevalence rates pre-fortification and
post-fortification.
Inclusion criteria: (1) Infants registered in
one of the 23 states affiliated to the Na-
tional Birth Defects Prevention Network,
(2) with one of the selected birth defects
(anencephaly, spina bifida without anen-
cephaly, common truncus, transposition of
the great arteries, tetralogy of Fallot, ventri-
cle septal defects, cleft palate only, cleft lip
without cleft palate, pyloric stenosis, renal
agenesis, bladder exostrophy, obstructive
genitourinary defects, reduction defects of
Outcomes: Prevalence rate of congenital II-2
abnormalities (PR) (N per 10000 live births)
pre- and post-fortification.
Results: PR combined with availability of
prenatal screening:
- Anencephaly PR=0.84; 95%CI 0.76 – 0.94.
- Spina Bifida PR=0.85; 95%CI 0.69-0.97.
- Common truncus PR=0.88; 95% CI 0.72-
1.08.
- Transposition of the great arteries
PR=0.88; 95% CI 0.81-0.96.

95
the upper and lower limbs, omphalocele,
Down syndrome), (3) live born or born after
pregnancy terminations in 8 states.
Study population: Total births n=9,729,763;
cases n=96,087.
Baseline characteristics: not reported.
Loss to follow-up: Not applicable.
Setting: 23 states in the USA.
- Tetralogy of Fallot PR=0.96; 95% CI 0.88-
1.04.
- Ventricular Septum Defect PR= 0.97;
95%CI 0.94-1.00.
- Cleft Palate PR 0.88; 95% CI 0.82-0.95
- Cleft lip and/or palate PR= 0.95; 95% CI
0.90-1.00
- Pyloric stenosis PR= 0.95; 95% CI 0.90-0.99
- Renal agenesis PR= 0.92; 95% CI 0.84-1.01
- Bladder exostrophy PR=1.13; 95% CI 0.82-
1.55.
- Obstructive genitourinary defects PR
=1.12; 95% CI 1.07-1.16.
- Upper limb reduction defects PR = 0.89
95% CI 0.80-0.99.
- Lower limb reduction defects PR= 0.97
95%CI 0.84-1.11).
- Omphalocele PR=0,79; 95%CI 0.66-0.95.
- Down syndrome PR=1,06; 95%CI 1,01-
1,11.

Review of preconception lifestyle interventions

4
 APPENDIX TABLE 2 (continued)
Intervention
Botto 200652 Aim: To evaluate international rates and
trends of 14 major defects in areas with
official recommendations to fortificate or
where fortification occurs.
Intervention: Folic acid national policy of
recommendations or actual food fortifi-
cation. This varied in type and timing per
country.
96
Yazdy 200756 Aim: To study the impact of folic acid fortifi-
cation on orofacial clefts.
Intervention: Folic acid fortification of grain
is mandatory as of 1998.
de Wals 200754 Aim: To assess changes in the prevalence of
before and after folic acid fortification.
Intervention: Folic acid fortification of grain
is mandatory as of 1998.
Methods
Design: Multicentre cohort study.
Recruitment: Population based retrospec-
tive surveillance data through 2003 on ma-
jor birth defects from 15 population-based
registries. Patient sampling: pre-recom-
mendation or fortification selected birth
defects vs. post-recommendation or forti-
fication selected birth defects.
Data collection/ follow-up: Collection of
rates and trends for 14 birth defects.
Design: Cohort study.
Recruitment: Population based retro-
spective from US birth certificates data.
Patient sampling: pre-fortification (1/1990-
12/1996) vs. post-fortification (10/1998-
12/2002).
Follow-up/ data collection: Comparing the
prevalence of orofacial clefts among births
before and after fortification.
Design: Cohort study.
Recruitment: Population based retro-
spective from 1993 to 2002 from multiple
sources ranging from provincial databases,
registry databases and medical records.
Patient sampling: pre-fortification selected
neural tube defects registered before 1997
vs. post-fortification selected neural tube
defects registered after 2000.
Data collection/ follow-up: Collection of
Population and setting
In-exclusion criteria: Unknown outcomes
of pregnancies and subjects with occult
spinal dysraphism, including spina bifi-
da occulta, thickened filum terminale,
diastematomyelia, caudal regression
syndrome, intradural lipoma, lipomenin-
gomyelocele, split notochord, and other
forms of myelodysplasia were excluded.
Study population: Total births n=1.5 mil-
lion yearly (from Europe n=1,000,000; from
Canada and the USA n=370,000; from Aus-
tralia n=87,000). Baseline characteristics:
not reported.
Loss to follow-up: Not applicable.
Setting: 15 registries participating in Eu-
rope, Australia, Canada and the USA.
In-exclusion criteria: Infants born with oro-
facial clefts were included; births between
1997 and 1998 were excluded.
Study population: N=38,232.
Baseline characteristics: Not specified.
Loss to follow-up: Not applicable.
Setting: 45 states and the district of Colum-
bia, USA.
In-exclusion criteria: All live births, still-
births and terminated pregnancies diag-
nosed with neural tube defects among
women residing in seven of the ten Ca-
nadian provinces were included. Subjects
with occult spinal dysraphism, including
spina bififda occulta, thickened filum
terminale, diastematomyelia, caudal re-
gression syndrome, intradural lipoma,
lipomeningomyelocele, split notochord,
Strength of
Outcomes evidence
Outcomes: Decrease in the prevalence of II-2
selected birth defects (live births, termina-
tions), including NTDs, per 1000 births for
the time period before and after the year
of introduction of national policy of recom-
mendations or fortification.
Results:
- Significant changes in trends were seen
for neural tube defects in areas with forti-
fication but not in areas with supplemen-
tation recommendations alone: varying
decrease from 10 to 35% in the data of Aus-
tralia (PPR=0.90; 95% CI 0.82-0.99), Western
Australia (PPR=0.70; 95% CI 0.61-0.81); Can-
ada (PPR=0.71; 95% CI 0.53-0.94); and USA
(PPR=0.65; 95% CI 0.56-0.76).
- For other major birth defects, there was
an overall lack of major trend changes after
recommendations or fortification.
Outcomes: Association between folic acid II-2
fortification and orofacial clefts.
Results:
Orofacial clefts declined following folic
acid fortification: PR=0.94; 95% CI 0.92-
0.96. The decline occurred in non-Hispanic
Whites, non-smokers, and women who
received prenatal care in the first trimester.
Outcomes: Decrease in the prevalence of II-2
neural tube defects per 1,000 births (live/
stillborn and terminated pregnancies) after
fortification.
Results:
- The prevalence of neural tube defects
decreased from 1.58/1,000 births before
fortification to 0.86/1,000 births during
the full-fortification period, 46% reduction;
95% CI 40-51.

Intervention
V PROGRAMS
Individual Interventions
Czeizel 199929 Aim: To evaluate the effect of ten years pre-
conception care.
Intervention: A check-up of reproductive
health, three months preparation for con-
ception (smoking cessation, alcohol drink-
ing, drug and medication use) and better
protection in early pregnancy.
97
Lumley 200630 Aim: Assessment of the effect of pre-preg-
nancy information, advice and counselling
on birth weight on subsequent pregnancy
outcomes.
Intervention: The intervention group re-
ceived a home visit with counselling (social
health and lifestyle problems/ timing of
pregnancy/ family history/ immunization/
referrals for health problems) and a re-
minder card. The comparison group only
received a visit about postpartum experi-
ences.
Review of preconception lifestyle interventions
Methods
annual incidence rates of neural tube de-
fects pre- and post-fortification.
Design: Cohort study.
Recruitment: Prospectively during 1984-
1994. Women were recruited by physicians,
midwives, nurses and social workers.
Data collection/ follow-up: Four visits to
a preconception care clinic (of which the
third and fourth occur during pregnancy)
with laboratory testing and data collection
of pregnancy outcome by birth certificate.
Design: Randomized controlled trial.
Recruitment: Women were recruited at
the Maternal and Child Health Centres be-
tween 1982 and 1994.
Data collection/ follow-up: Data on the
subsequent pregnancy outcome was col-
lected by visit, telephone and mail.
Population and setting
and other forms of myelodysplasia were
excluded.
Study population: Total births n=1.9 mil-
lion, cases n=2446.
Baseline characteristics: not reported.
Loss to follow-up: Not applicable.
Setting: Seven provinces in Canada.
In- exclusion criteria: Voluntary of
non-pregnant and non subfertile women.
Study population: N=8,837 of which 6,060
confirmed pregnancies. Age: mean 25.8
years ± 3.4, 85% was primiparous, 60% had
the highest class of education.
Loss to follow-up: None reported.
Setting: Risk screening by qualified nurses
in an outpatient clinic in Hungary.
In- and exclusion criteria: Postpartum
women attending government funded
local Maternal and Child Health centres, in
a community setting with high risk of poor
birth outcomes (due to socioeconomic
reasons) Women attending specialist care
were excluded.
Study population: N= 786 (intervention
group n=392; comparison group n=394).
The two groups did not differ in baseline
characteristics.
Loss to follow-up: 392/777 and 394/802
women were analysable.
Setting: In Melbourne, Australia in a newly
established pre-pregnancy walk-in service
and the home of the participants. Counsel-
ling by midwives.
4
Strength of
Outcomes evidence
- The decrease was greater for spina bifida
(53%) than for anencephaly and encepha-
locele (38% and 31%, respectively).
Outcomes: Smoking cessation (verified by II-2
urine cotinine).
Results: At initial consultation 17.9% was a
smoker vs. 12.4% 3 months later.
Outcomes: Primary outcome: difference in I-A
birth weight compared to index pregnan-
cy. Secondary outcomes: gestational age at
birth, low birth weight (<2500gram), birth
weight <10th percentile, perinatal deaths
and birth defects.
Results:
- Birth weight was significantly 97.4 gram
lower among infants in the intervention
group.
- No significant differences were found for
preterm birth (OR 1.44; 95% CI 0.73-2.91),
low birth weight (OR 1.85; 95% CI 0.91-
3.91) or birth weight <10th percentile (OR
1.14; 95% CI 0.55-2.38).
- Compared to the controls, the interven-
tion group had more preterm births <32
weeks (10 vs. 1), more birth weights <2000
g (16 vs. 2), and more perinatal deaths due
to birth anomalies (5 vs. 2).
 APPENDIX TABLE 2 (continued)
Intervention
Elsinga 200831 Aim: Assessment of preconceptional
health counselling.
Intervention: The intervention group re-
ceived counselling after risk assessment.
The control group received standard care
as provided their general practitioner.
98
Ockhuijsen 201132 Aim: To assess efficacy of interventions
aimed at altering behavior patterns relat-
ing to smoking and obesity in a precon-
ception care service integrated in an IVF
programme.
Intervention: Consisted of counselling (not
specified) during visits and by telephone.
Williams 201233 Aim: To assess the effect of preconception
care on positive maternal behaviors before
and during pregnancy.
Methods
Design: Randomized controlled trial.
Recruitment: Prospectively during 2000-
2003. Recruited by annual invitations sent
by general practitioners to the eligible
sample of patients in their practice.
Randomization: At the level of general
practices.
Data collection/ follow-up: Risk assessment
questionnaire was sent at baseline and
within two months after delivery.
Design: Cohort study.
Recruitment: Prospectively from 2007 -
2008.
Data collection/ follow-up: Self-reported
weight and smoking status and measured
weight. Follow-up by telephone call or visit
every 4 weeks. The length of follow-up was
up to 1 year.
Design: Cross-sectional study.
Recruitment: Retrospectively during 2004
– 2008. Women were recruited by sampling
Population and setting
In- exclusion criteria: Women aged 18-40
contemplating pregnancy within one year;
in cases were invitation was undesirable
women were not invited.
Study population: The intervention group:
n=211 women and 150 pregnancies; the
control group: n= 422 women and 1,914
pregnancies.
Baseline characteristics (intervention vs.
comparison group): age 20-30 (26,8% vs.
28,5%), age 30 ->40 (73,2% vs. 71,5%);
country of birth in the Netherlands (94,7%
vs. 87,8%); educational level high (45,1%
vs. 37,6).
Loss to follow-up: 82% of the question-
naires obtained after pregnancy in the in-
tervention group were analysable vs. 68%
in the control group.
Setting: General practitioners practices, the
Netherlands.
In- exclusion criteria: Couples on the wait-
ing list for an IVF treatment who had visit-
ed the preconception clinic and were able
to read Dutch.
Study population: N=101. Baseline charac-
teristics: 30 women had a BMI >30 kg/m2
(mean value of 33.8; SD 3.6); 23 women
smoked.
Loss to follow-up: n=25 (obese group
n=15; smoking group n=10) and were ex-
cluded.
Setting: Nurses who were trained for pre-
conception care counselling and had at
least 3 months experience at an IVF unit,
university hospital, the Netherlands.
In- exclusion criteria: Women that had de-
livered a live birth 2-6 months ago. Women
that reported drinking or smoking during
Strength of
Outcomes evidence
Outcomes: I-a
Smoking**, alcohol consumption*,
binge drinking*, start of folic acid use*,
adverse pregnancy outcomes (prema-
ture birth (<37 weeks); low birth weight
(<2500grams), small for gestational age
(growth <p2,3) and congenital anomalies.
* up to 3 months before the pregnancy
** in or up to 3 months before pregnancy
Results:
Intervention group vs. comparison group:
- Smoking cessation**:18% vs. 10% unad-
justed OR=3,04; 95%CI 0,95-9,69.
- Folic acid use*: 86% vs. 53%; adjusted
OR=4,93; 95%CI 2,81-8,66.
- Alcohol consumption: * 32% vs. 45%; ad-
justed OR=1,79 95%CI 1,08–2,97.
- Percentage of adverse outcome: 16,2% vs.
20,2%; OR 0.77; 95%CI 0.48-1.22.
Outcomes: Mean weight reduction (self-re- II-2
ported and weighed combined) in kg and
smoking cessation.
Results:
Weight loss:
- 15/30 women (50%) lost weight (mean
6.1 kg; SD 3.6); 11 of these 15 women
reached the goal of losing 5% or more of
their original weight.
Smoking cessation:
- 7/23 women (30%) quit smoking
- 6/23 women (26%) reduced the number
of cigarettes.
Outcomes: Prepregnancy daily multivita- II-2
min consumption, first trimester entry into
prenatal care, smoking cessation (not

Intervention
Intervention: Any form of contact with a
health care worker preconceptionally to
prepare for a healthy pregnancy and baby
compared to no contact with a health care
worker to prepare for pregnancy.
Group interventions
Hillemeier 200840 Aim: To assess the efficacy of a preconcep-
99
tional health programme.
Intervention: The intervention group un-
derwent six 2-hour group sessions spread
out over a 12-week period (topics were
managing stress, physical activity, nutrition
(including folic acid supplementation), pre-
venting gynaecologic infection, tobacco
exposure, and alcohol use.) The compar-
ison group did not receive the group ses-
sions.
Review of preconception lifestyle interventions
Methods
of state birth certificates.
Data collection/ follow-up: Data from the
Pregnancy Risk Assessment Monitoring
System (PRAMS) was used; additional data
was retrieved by mailed questionnaires.
Non-responders were contacted by tele-
phone.
Design: Randomized controlled trial.
Recruitment: Prospectively. Time frame un-
clear. Women were recruited via triangular
community based approach with active
(personal communication) and passive
recruitment (media, mailings, posters and
flyers).
Randomization: women were randomized
after baseline risk assessment, stratified
by site.
Data collection/ follow-up: Participants re-
ceived a baseline and follow-up health risk
assessment at 14 weeks (questionnaires,
anthropometric measurements, and bio-
markers).
Population and setting
follow-up that did not report this at base
line were excluded.
Study population: N= 30,481 women of
which 9,457 received preconception care.
Baseline characteristics: the population
that received preconception care con-
tained more women >20 years of age,
more non-Hispanic white and black wom-
en, years of education ≥12 years, that were
married, that were privately insured, have a
normal weight, have a prior live birth, have
a prior intended pregnancy, have diabetes
of a poor prior birth outcome.
Loss to follow-up: Response rates ranged
from 72 to 89%.
Setting: USA (Maine, New Jersey, Utah,
Vermont.
In- exclusion criteria: (1) Residence with-
in the 28 county target study region; (2)
women aged 18-35; (3) not pregnant at
time of enrolment; (4) capable of becom-
ing pregnant; (5) English speaking.
Study population: N=692 (intervention
group n=473; comparison group n=219).
Baseline characteristics intervention vs.
comparison group: married or living with
partner 59% vs. 48%; mean age 26.52 years
(SD 5.02) vs. 24.74 years (SD 4.64); high
school education 36% vs. 31%; race White/
non-Hispanic 92% vs. 91%, Other 8% vs.
9%; poor poverty status 27% vs. 29%.
Loss to follow-up: 47% in the intervention
group and 50% in the comparison group
did not attend the follow-up risk assess-
ment. The analytic sample consisted of 362
women (intervention group n=252; com-
parison group n=110).
Setting: Community based; in 15 low-in-
come rural communities, Central Pennsyl-
vania.
4
Strength of
Outcomes evidence
smoking on an average day within the pri-
or three months to pregnancy) and cessa-
tion of alcohol consumption (not drinking
with the last three months prior to preg-
nancy).
Results: Receipt of preconception care was
significantly associated with:
- Daily consumption of multivitamins one
month before pregnancy (AOR 4.35; 95%
CI 4.00-4.73).
- Cessation of drinking during the three
months before pregnancy (AOR 1.34; 95%
CI 1.16-1.54).
Outcomes: Behavioral change related pri- I-a
marily to nutrition and physical activity.
Results:
Statistically significant behavior changes
included greater likelihood of:
- Reading food labels to identify nutritional
values (OR 2.264; p-value 0.001).
- Using a daily multivitamin that contains
folic-acid (OR 6.595; p-value <0.001).
- Meeting recommended levels of physical
activity levels (OR 1.867; p-value 0.019).
Significant dose effects were found for:
- Reading food labels (OR 1.161; p-value
0.015)
- Daily use of multivitamin with folic-acid
(OR 1.448; p-value <0.001).
- Anthropometric measurements (BMI,
waist circumference, and blood pressure)
and biomarkers (serum glucose, HDL cho-
lesterol, and total cholesterol) were not
significantly different in pre- and post-anal-
ysis.
 APPENDIX TABLE 2 (continued)
Intervention
Weisman 201141 Aim: To assess the long-term effects of the
Strong Healthy Women intervention.
Intervention: See Hillemeier 2008.
100
Methods
Design: Follow up of the RCT Hillemeier
2008
Data collection/ follow-up: Telephone in-
terviews 6- and 12- months after group
sessions in Hillemeier 2008. Birth records
from women who gave birth to singletons
during the 12-months follow-up period.
Population and setting
In- exclusion criteria: see Hillemeier
2008.40
Study population:
The analytic sample n=362 from the orig-
inal trial were included. Data at 6 months
reported on n=315; at 12 months reported
on n=302. Data on 45 births were collect-
ed.
Loss to follow-up: At six-month n=47; at
twelve months n=60.These women were
excluded.
Strength of
Outcomes evidence
Outcomes: (1) maintenance of significant I-a
pretest-posttest changes; (2) impact on
weight, BMI and daily folic acid use, and (3)
impact on weight gain during pregnancy.
Results:
At 12 months follow-up women of the in-
tervention group were more likely:
- To use multivitamin with folic acid
daily (OR 2.15; 95% CI 1.19-3.88).
- To have a lower weight (OR -4.33; 95% CI
-8.16- -0.49).
- To have a lower BMI (OR -0.75; 95% CI
-1.39- -0.11).
- Intervention effect on physical activity,
consumption of fruit and vegetables was
not maintained during the follow-up pe-
riods (OR 1.00; 95% CI 0.57-1.76, OR 0.68;
95% CI 0.39-1.21, OR 0.99; 95% CI 0.57-
1.71).
- Intervention effect on reading food labels
for nutritional values decreased between
the 6- and 12-month follow-up (OR 1.97;
95% CI 1.07-3.65 and OR 0.70; 95% CI 0.40-
1.23).
4

Review of preconception lifestyle interventions

101
 PART II

2
DESIGNING AN
INTERVENTION STRATEGY
THE HEALTHY PREGNANCY
4 ALL STUDY: DESIGN AND
COHORT PROFILE
S. Denktaş, J. Poeran, L.C. de Jong-Potjer, S.F. van Voorst,
A.A. Vos, A.J.M. Waelput, E. Birnie, G.J. Bonsel, E.A.P. Steegers
BMC Pregnancy and Childbirth 2014, 14:253
ABSTRACT
Background: Promotion of healthy pregnancies has gained high priority in the Netherlands
because of the relatively unfavourable perinatal health outcomes. In response a nationwide study
Healthy Pregnancy 4 All was initiated. This study combines public health and epidemiologic
research to evaluate the effectiveness of two obstetric interventions before and during pregnancy:
(1) programmatic preconception care (PCC) and (2) a systematic antenatal risk assessment
(including both medical and non-medical risk factors) followed by patient-tailored multidisciplinary
care pathways. In this paper we present an overview of the study setting and outlines. We describe
the selection of geographical areas and introduce the design and outline of the preconception care
and the antenatal risk assessment studies.
Methods/design: A thorough analysis was performed to identify geographical areas in which
adverse perinatal outcomes were high. These areas were regarded as eligible for either or both sub-
studies as we hypothesised studies to have maximal effect there. This selection of municipalities was
based on multiple criteria relevant to either the preconception care intervention or the antenatal
risk assessment intervention, or to both. The preconception care intervention was designed as
a prospective community-based cohort study. The antenatal risk assessment intervention was
designed as a cluster randomised controlled trial – where municipalities are randomly allocated to
intervention and control.
Discussion: Optimal linkage is sought between curative and preventive care, public health,
government, and social welfare organisations. To our knowledge, this is the first study in which
these elements are combined.

106
BACKGROUND
Perinatal mortality rates in the Netherlands are high and decline slower than in other European
countries.1-3 Furthermore, an inequality in adverse perinatal outcomes is seen as more risks and
a higher risk load for adverse outcomes were found for women living in socially deprived areas.4
Population-based cohort studies, e.g. the Generation R and ABCD studies have contributed to
our knowledge of various health problems in pregnancy and childhood and their lasting impact
on health in later life.5,6 Studies using a large national Dutch database (The Netherlands Perinatal
Registry) showed increased adverse pregnancy outcome in large urban areas, in particular in
deprived neighborhoods.7,8 Analyses of this database provided recognition that four specific
morbidities precede perinatal mortality in 85% of cases, the so-called ‘Big4’ morbidities.9,10
These are: congenital anomalies (list defined), preterm birth (<37th week of gestation), small for
gestational age (SGA, birth weight <10th percentile for gestational age) or low Apgar score (<7, 5
minutes after birth).
Taking prior research into account, a nationwide study focusing on deprived areas with a higher
than average perinatal mortality and morbidity rate was designed. Our strategy was to perform
a thorough epidemiological analysis to identify areas in which interventions would theoretically
5
have the highest impact in improving perinatal health.

Design of the HP4ALL study

HEALTHY PREGNANCY 4 ALL
With the support of the Ministry of Health and Welfare a nationwide study called ‘Healthy Pregnancy
4 All’ (HP4All), was initiated. Several municipal pilot studies in the city of Rotterdam provided its
framework.11 The main objective of HP4All is to evaluate the effectiveness of the interventions and
their associated preventive strategies in either the preconception period or the antenatal period
to reduce adverse pregnancy outcome. Accordingly, two sub-studies are designed: a population-
based prospective cohort study focusing on the effectiveness of customized preconception care
(PCC) and a systematic antenatal risk assessment score-card including both medical and non-
medical risk factors followed by patient-tailored multidisciplinary care pathways.12,13
The rationale of the PCC sub-study originates from increasing evidence showing the critical
influence of embryonic development and placentation during early pregnancy on pregnancy
outcome.14-16 Risks influencing this early pregnancy phase can be modified optimally in the
preconception period.16-18 The Dutch Health Council recommended (2007) to integrate general
PCC in the health care system.19 The Minister of Health, however, advised to evaluate the utilization
and effectiveness of PCC for high risk groups first, before collective reimbursement of PCC in Dutch
obstetric care would be (re)considered.
The second sub-study concerns a cluster randomized controlled trial, focusing on the early
detection of risks for adverse pregnancy outcomes with a score card including both medical and
non-medical risks. The unique Dutch system of obstetric care system has three risk-based levels of
care: primary care (indicated for low risk pregnancies and deliveries, provided by independently
practicing midwives), and secondary/tertiary care (indicated for high risk pregnancies, provided

107
by obstetricians).20 As the level of care depends on the distinction between low risk and high risk
pregnancies, antenatal risk assessment is an important part of Dutch obstetric care.20 Although
social deprivation has been shown to contribute to adverse perinatal health in the Netherlands,
standard risk assessment does not include the assessment of non-medical risks of perinatal
health.4,20-23 In addition, subsequent patient-tailored pathways are lacking. Therefore, in the new
antenatal risk assessment tool (‘R4U score card’) both medical and non-medical risk factors are
explicitly taken into account as part of the HP4All study.
The aim of this paper is to present an overview of the HP4All study. Below, we first describe the
selection of geographical areas most eligible for the interventions. Next we introduce the design of
the preconception care and the antenatal risk assessment sub-studies.

METHODS/DESIGN
Identification and selection of the eligible geographical areas for the interventions
The first step was the identification of the geographical units in which the aforementioned sub-
studies would preferably be carried out. We used a national Geographic Information System (GIS) to
divide The Netherlands into 62 municipalities, being the 50 municipalities with > 70.000 inhabitants
and the 12 provinces (excluding the 50 previously selected municipalities). The second step
involved the selection of municipalities in which to carry out the sub-studies, based on multiple
criteria which are relevant to either the preconception care intervention or broadened antenatal
risk assessment.
Of the 50 cities with >70.0000 inhabitants, we selected municipalities according to socio-
demographic parameters associated with high risk load (maternal age, parity, ethnicity, and
socioeconomic status) and perinatal outcome data (overall ‘Big4’ and perinatal mortality
prevalence). Before the municipalities could be selected, specific parameters that make delivery of
PCC or antenatal risk assessment relevant were applied.
For the PCC sub-study these criteria were (1) proportion of women having their first antenatal
booking visit at ≥14 weeks of gestational age, and prevalences of (2) congenital anomalies and of
(3) SGA. The moment of the first antenatal booking is important because it is a condition for timely
intervention upon present risk factors. The effectiveness of these interventions is larger in an early
fetal stage. Congenital anomaly and SGA prevalences are considered to be indicative for a region’s
periconceptional health status.
For the antenatal risk assessment sub-study, additional criteria were (1) overall perinatal
mortality rates, (2) perinatal mortality amongst women with ‘Big4’ pregnancies (‘case-fatality’), and
(3) prevalence of SGA and prematurity. For each specific indicator we present the absolute rate, the
standardised rate and the so-called inequality-rate, the latter being expressed as the relative risk of
the outcome for low SES (socioeconomic status) pregnant women compared to high SES pregnant
women, after direct standardisation for maternal age, parity and ethnicity. Standardisation is
needed because a region with, e.g. a high number of non-Western women or a high number of
teenage pregnancies will generally have a higher prevalence of adverse perinatal outcomes.

108
DATA SOURCES
The division of The Netherlands into 62 municipalities was based on 4-digit postal codes areas.
Data were provided by the Falk company (www.falk.nl), the National Public Health Authority,
and the Statistics Netherlands organisation (CBS, www.cbs.nl). Information on socioeconomic
status (SES, determined in 2006) per postal code area was obtained from the Social and Cultural
Planning Office (SCP, www.scp.nl). Data on pregnancy and perinatal outcome were derived from
The Netherlands Perinatal Registry (2000–2008). This database contains information of more than
97% of all pregnancies in The Netherlands.24 The data are routinely collected by 94% of midwives,
99% of gynaecologists and 68% of paediatricians including 100% of Neonatal Intensive Care Unit
paediatricians.24
Table 1 shows the demographic characteristics of the so-called ‘G4-cities’, i.e. the four largest
cities: Amsterdam, Rotterdam, The Hague, Utrecht, and the rest of the Netherlands. Compared to
the rest of The Netherlands, the ‘G4’-cities have a larger proportion of non-Western women (43% vs.
11.3%), more teenage pregnancies (2.8% vs. 1.5%), and more women in low SES neighbourhoods
(59.2% vs. 19.0%). Considerably more women live in deprived neighbourhoods (32.5% vs. 1.3%)
and the overall adverse perinatal outcome is worse in ‘G4-cities’, as illustrated by a ‘Big4’ prevalence
5
of 20.5% compared to 18.1%.

Design of the HP4ALL study

Perinatal mortality and ‘Big4’ prevalence
Figures 1 and 2 illustrate the geographical distribution (50 municipalities and 12 provinces) of
perinatal mortality rates, and the prevalence rate of ‘Big4’ (per 1,000), respectively. Various shades of
red represent the different prevalence classes, the darker the shade the more prevalent the adverse
outcome. The classes are based on the distribution of the rates: the middle three classes comprise
95% (2 standard deviations) of the outcome levels; the middle class comprises 68%. Both figures
show large geographical inequalities in adverse perinatal outcomes on the national level.

Comparison municipalities
Additionally, we compared these outcomes across areas after direct standardisation for population
differences by maternal age, parity, ethnicity, and SES.25 Standardisation is needed because a region
with, e.g. a high number of non-Western women or a high number of teenage pregnancies will
generally have a higher prevalence of adverse perinatal outcomes.
Tables 2 and 3 show the socio-demographic parameters and the specific criteria for the PCC
and the antenatal risk assessment sub-studies. For each specific indicator we present the absolute
rate (ABS), the standardized rate (STND) and the inequality-rate (INEQ, the relative risk of the
standardised outcome for low SES pregnant women compared to high SES pregnant women).
Next, to facilitate comparisons, we assigned decile scores to regions, varying from one (the region
is one of the 10% areas with best outcomes) to 10 (the region belongs to the 10% worst outcomes).
The sum of the decile scores for the various indicators by region is shown in the last column
(‘RANK’); higher scores imply unfavourable ranking. Based on the sum of the decile scores for the
PCC sub-study (table 2), the municipalities have the most adverse outcomes, i.e. 1. The Hague; 2.

109
TABLE 1: D
 emographic characteristics of the study population by yes/no ‘G4-cities’
(the four largest cities) with percentages in brackets

G4-CITIES NETHERLANDS MINUS TOTAL

G4-CITIES
No.of pregnancies during study period 245445 (100.0) 1338420 (100.0) 1583865 (100.0)
Parity
Primiparous 121592 (49.5) 607953 (45.4) 729545 (46.1)
Multiparous 123853 (50.5) 730467 (54.6) 854320 (53.9)
Ethinicity
Western 139786 (57.0) 1186772 (88.7) 1326558 (83.8)
Non-Western 105659 9(43.0) 151648 (11.3) 257307 (16.2)
Maternal age
< 20 years 6987 (2.8) 19861 (1.5) 26848 (1.7)
20-24 years 34864 (14.2) 127013 (9.5) 161877 (10.2)
25-29 years 61354 (25.0) 395138 (29.5) 456492 (28.8)
30-34 years 85444 (34.8) 535927 (40.0) 621371 (39.2)
≥ 35 years 56796 (23.1) 260481 (19.5) 317277 (20.0)
Socioeconomic ‘status score’
<p20 145367 (59.2) 254607 (19.0) 399974 (25.3)
p20-p80 58641 (23.9) 853074 (63.7) 911715 (57.6)
>p80 41437 (16.9) 230739 (17.2) 272176 (17.2)
Neighbourhood
Non-deprived 165658 (67.5) 1320392 (98.7) 1486050 (93.8)
Deprived 79787 (32.5) 18028 (1.3) 97815 (6.2)
Perinatal outcomes**
Congenital anomalies 5233 (2.1) 33159 (2.5) 38392 (2.4)
Preterm birth 15673 (6.4) 81646 (6.1) 97319 (6.1)
Small for gestational age 27724 (11.3) 125175 (9.4) 152899 (9.7)
Apgar score <7 3385 (1.4) 14818 (1.1) 18203 (1.1)
(5 minutes after birth)
Any Big4** 50267 (20.5) 242697 (18.1) 292964 (18.5)
Fetal mortality† 1478 (0.6) 6718 (0.5) 8196 (0.5)
Intrapartum mortality 458 (0.2) 2126 (0.2) 2584 (0.2)
Neonatal mortality†† 761 (0.3) 3547 (0.3) 4308 (0.3)
Perinatal mortality‡ 2697 (1.1) 12391 (0.9) 15088 (1.0)

** Individual ‘Big4’ morbidities do not add up to ‘any Big4’.

as women can have >1 ‘Big4’ morbidity.
† From 22 weeks of gestational age.
†† 0–7 days postpartum.
‡ Total of fetal, intrapartum and neonatal mortality.

110
FIGURE 1: A
H5
 bsolute prevalence of perinatal
mortality per 1000 births.
F1

Design of the HP4ALL study

FIGURE 2: A
 bsolute prevalence of ‘Big4’
morbidities per 1000 births.

111
TABLE 2: S
 election criteria* for the preconception care experiment with scoring in deciles; the higher
deciles represent a more likely qualification for inclusion.

1ST ANTENATAL CONGENITAL

DEMOGRAPHICS SGA RANK
BOOKING ≥14W ANOMALIES
% AGE NW LOW
# CITIES ABS STND INEQ ABS STND INEQ ABS STND INEQ
PREG <20 ETHN SES
1 Amsterdam 10 8 10 10 10 10 3 3 2 7 8 6 9 96
2 Rotterdam 10 10 10 10 10 10 3 6 7 4 10 9 6 105
3 Den Haag 9 10 10 10 10 10 2 9 8 4 10 9 8 109
4 Utrecht 9 3 10 6 10 10 4 10 10 7 3 2 7 91
5 Eindhoven 8 7 9 7 9 9 6 8 9 8 9 9 5 103
6 Tilburg 8 8 9 9 5 4 10 4 4 5 10 10 3 89
7 Almere 8 7 10 3 10 9 1 7 7 6 9 8 8 93
8 Groningen city 7 9 5 9 2 2 5 2 2 4 5 3 5 60
9 Breda 7 6 6 5 3 1 9 9 9 3 6 7 4 75
10 Nijmegen 7 5 6 9 3 3 9 4 5 6 8 8 6 79
11 Enschede 6 8 8 10 4 4 2 5 5 3 9 7 6 77
12 Apeldoorn 6 5 3 2 6 7 4 1 1 9 5 4 10 63
13 Haarlem 7 3 7 6 8 7 3 1 2 7 4 4 7 66
14 Arnhem 6 9 9 8 8 5 7 6 6 3 7 7 5 86
15 Zaanstad 6 4 8 6 7 7 1 2 3 2 5 4 8 63
16 Amersfoort 7 2 7 4 9 9 7 5 6 6 3 2 4 71
17 Haarlemmermeer 7 1 4 1 4 5 4 1 1 2 2 2 7 41
18 's-Hertogenbosch 5 3 3 4 1 2 10 9 9 4 8 8 4 70
19 Zoetermeer 5 6 8 3 1 1 6 4 4 1 7 6 10 62
20 Zwolle 6 7 3 4 2 3 7 2 1 7 2 1 10 55
21 Maastricht 4 9 4 6 4 3 10 10 10 10 10 10 1 91
22 Dordrecht 6 10 9 7 9 8 3 2 1 3 7 7 8 80
23 Leiden 5 4 7 6 8 8 6 8 7 10 6 5 3 83
24 Emmen 4 6 1 10 4 5 10 2 2 7 6 4 9 70
25 Ede 5 6 3 5 5 6 6 7 8 2 1 1 5 60
26 Venlo 3 7 8 7 3 2 8 6 6 5 9 10 1 75
27 Westland 4 1 1 1 5 7 6 10 10 8 1 1 10 65
28 Deventer 5 6 6 8 7 8 7 7 7 2 7 7 2 79
29 Delft 3 7 9 9 7 5 7 10 10 10 5 5 6 93
30 Sittard-Geleen 3 8 3 7 1 2 10 5 4 4 9 8 1 65
31 Leeuwarden 4 10 4 9 5 4 8 8 8 2 5 3 10 80
32 Alkmaar 4 4 6 5 6 6 8 5 5 10 2 2 2 65
33 Heerlen 2 10 5 10 3 4 8 10 10 3 10 10 2 87
34 Helmond 5 5 7 6 6 5 4 8 8 5 9 10 1 79
35 Hilversum 1 5 5 3 9 9 1 1 1 8 3 5 1 52
36 Súdwest Fryslân 3 5 1 8 2 2 9 2 2 10 1 1 3 49
37 Amstelveen 2 1 8 2 8 8 1 1 1 10 2 1 10 55
38 Hengelo 4 6 4 7 5 6 1 4 3 1 4 4 5 54
39 Purmerend 2 4 6 4 9 10 1 3 5 1 4 6 9 64
40 Roosendaal 2 5 9 1 2 1 8 9 9 8 8 10 1 73
41 Oss 2 2 4 3 1 1 7 5 4 9 10 10 2 60
42 Schiedam 1 10 10 10 10 10 2 7 6 4 10 9 7 96

112
TABLE 2 (continued)

1ST ANTENATAL CONGENITAL

DEMOGRAPHICS SGA RANK
BOOKING ≥14W ANOMALIES
% AGE NW LOW
# CITIES ABS STND INEQ ABS STND INEQ ABS STND INEQ
PREG <20 ETHN SES
43 Spijkenisse 1 9 7 4 3 2 5 3 3 1 6 9 4 57
44 Leidschendam-Voorburg 2 2 7 3 8 7 5 5 4 9 3 5 5 65
45 Alphen a/d Rijn 1 2 5 1 4 4 9 7 8 1 4 4 6 56
46 Almelo 3 8 5 8 2 3 1 1 1 9 7 6 1 55
47 Vlaardingen 1 8 10 5 7 4 8 6 5 9 8 8 4 83
48 Gouda 3 3 8 8 3 1 9 1 3 3 4 3 3 52
49 Middelburg 1 9 4 7 6 6 4 8 6 6 4 3 3 67
50 Vlissingen 1 10 6 5 8 6 5 6 8 1 8 9 3 76

# PROVINCES
51 Groningen 8 7 2 9 7 9 5 3 2 8 5 6 7 78
52
53
Friesland
Drenthe
9
9
4
3
1
1
8
5
9
6
9
8
3
6
10
4
10
4
8
2
2
3
3
5
9
8
85
64
5
54 Overijssel 9 1 1 2 5 7 2 3 3 6 1 2 9 51

Design of the HP4ALL study

55 Gelderland 10 2 2 2 1 3 3 10 9 9 2 3 6 62
56 Utrecht 10 1 3 1 2 3 5 9 9 5 1 1 7 57
57 Noord-Holland 10 1 2 2 7 8 2 6 6 5 1 1 8 59
58 Zuid-Holland 10 2 2 1 4 5 4 8 7 7 1 2 9 62
59 Zeeland 8 3 1 3 10 10 2 4 5 1 3 5 4 59
60 Noord-Brabant 10 1 2 1 1 1 9 7 7 5 6 7 2 59
61 Limburg 9 4 2 2 1 1 10 9 10 6 7 8 2 71
62 Flevoland 8 9 5 4 6 6 6 3 3 9 6 6 7 78

* ‘% PREG’: % Of pregnant women in the general population

* ‘AGE <20’: % Of teenage pregnancies
* ‘NW ETHN’: % Of pregnant women with a non-Western ethnicity
* ‘LOW SES’: % Of women in neighbourhoods with a socioeconomic status score <p20
* ‘ABS’: Absolute %
* ‘STND’: Standardised %
* ‘INEQ’: Inequality as measured by the relative risk of prevalences between women from neighbourhoods with soci-
oeconomic status score <p20 compared to >p80

113
TABLE 3: S
 election criteria* for the risk selection experiment with scoring in deciles; the higher deciles
represent a more likely qualification for inclusion.

PERINATAL PERINATAL MORTAL-

PERINATAL MORTAL-
DEMOGRAPHICS MORTALITY / BIG4 ITY / START LABOUR RANK
ITY / ALL WOMEN
MORBIDITIES IN PRIMARY CARE
% AGE PRIMI NW LOW
# CITIES ABS STND INEQ ABS STND INEQ ABS STND INEQ
PREG <20 ETHN SES
1 Amsterdam 10 8 10 10 10 8 6 9 8 7 7 7 5 8 113
2 Rotterdam 10 10 7 10 10 10 10 3 6 7 3 10 9 5 110
3 Den Haag 9 10 7 10 10 9 8 7 6 7 4 10 8 9 114
4 Utrecht 9 3 9 10 6 9 9 2 9 10 2 7 6 5 96
5 Eindhoven 8 7 9 9 7 5 5 4 2 2 2 9 8 6 83
6 Tilburg 8 8 7 9 9 8 8 6 4 5 8 9 9 3 101
7 Almere 8 7 4 10 3 8 10 3 5 8 3 6 7 7 89
8 Groningen city 7 9 10 5 9 7 9 1 8 9 3 2 1 7 87
9 Breda 7 6 6 6 5 3 4 7 2 4 6 7 8 3 74
10 Nijmegen 7 5 8 6 9 10 10 4 10 10 2 6 6 7 100
11 Enschede 6 8 5 8 10 9 9 4 8 6 3 9 8 3 96
12 Apeldoorn 6 5 4 3 2 8 8 8 9 8 8 3 4 10 86
13 Haarlem 7 3 9 7 6 4 6 8 5 6 9 3 2 7 82
14 Arnhem 6 9 10 9 8 9 4 9 9 6 8 5 2 8 102
15 Zaanstad 6 4 6 8 6 2 1 1 2 1 4 5 6 4 56
16 Amersfoort 7 2 6 7 4 10 10 5 10 10 7 1 1 8 88
17 Haarlemmermeer 7 1 5 4 1 4 3 7 7 6 7 1 1 6 60
18 's-Hertogenbosch 5 3 10 3 4 6 5 3 4 4 5 6 7 5 70
19 Zoetermeer 5 6 6 8 3 1 1 2 1 1 1 6 7 10 58
20 Zwolle 6 7 6 3 4 6 2 5 8 4 2 4 1 10 68
21 Maastricht 4 9 8 4 6 8 7 8 3 2 6 10 9 6 90
22 Dordrecht 6 10 4 9 7 2 1 3 2 1 5 7 4 10 71
23 Leiden 5 4 10 7 6 4 2 9 3 2 9 4 5 3 73
24 Emmen 4 6 4 1 10 2 2 1 3 3 1 8 6 10 61
25 Ede 5 6 1 3 5 7 4 9 9 5 10 1 3 2 70
26 Venlo 3 7 5 8 7 3 2 10 3 1 10 10 10 2 81
27 Westland 4 1 1 1 1 1 2 8 1 1 8 8 7 9 53
28 Deventer 5 6 6 6 8 9 9 3 7 5 4 9 10 3 90
29 Delft 3 7 8 9 9 1 1 5 1 1 1 10 10 8 74
30 Sittard-Geleen 3 8 9 3 7 3 1 7 1 1 9 9 9 1 71
31 Leeuwarden 4 10 9 4 9 5 5 10 5 5 10 5 5 5 91
32 Alkmaar 4 4 7 6 5 2 2 10 4 3 10 3 4 1 65
33 Heerlen 2 10 10 5 10 7 8 6 1 2 8 10 10 4 93
34 Helmond 5 5 4 7 6 5 4 8 4 3 10 8 8 2 79
35 Hilversum 1 5 10 5 3 7 5 2 10 8 6 3 3 4 72
36 Súdwest Fryslân 3 5 2 1 8 7 7 10 10 10 10 1 1 7 82
37 Amstelveen 2 1 3 8 2 1 1 10 7 5 9 1 1 10 61
38 Hengelo 4 6 3 4 7 5 7 5 6 6 7 4 4 4 72
39 Purmerend 2 4 8 6 4 2 3 9 5 4 9 7 9 5 77
40 Roosendaal 2 5 5 9 1 2 5 2 1 2 5 9 10 1 59
41 Oss 2 2 5 4 3 3 4 7 1 2 7 8 7 6 61

114
TABLE 3 (continued)

PERINATAL PERINATAL MORTAL-

PERINATAL MORTAL-
DEMOGRAPHICS MORTALITY / BIG4 ITY / START LABOUR RANK
ITY / ALL WOMEN
MORBIDITIES IN PRIMARY CARE
% AGE PRIMI NW LOW
# CITIES ABS STND INEQ ABS STND INEQ ABS STND INEQ
PREG <20 ETHN SES
42 Schiedam 1 10 9 10 10 10 10 9 6 4 8 5 1 4 97
43 Spijkenisse 1 9 8 7 4 10 8 6 9 8 4 6 7 7 94
44 Leidschendam-Voorburg 2 2 7 7 3 1 1 10 4 3 10 2 3 8 63
45 Alphen a/d Rijn 1 2 8 5 1 10 10 1 10 10 5 4 3 5 75
46 Almelo 3 8 3 5 8 1 3 2 2 5 1 6 6 1 54
47 Vlaardingen 1 8 7 10 5 7 10 3 6 10 3 10 10 2 92
48 Gouda 3 3 1 8 8 6 3 10 7 6 9 2 2 3 71
49 Middelburg 1 9 1 4 7 1 3 1 3 4 2 2 2 2 42
50 Vlissingen 1 10 4 6 5 6 9 1 4 7 1 8 10 1 73

# PROVINCES
51 Groningen 8 7 3 2 9 9 8 6 10 9 6 5 5 4 91 5
52 Friesland 9 4 2 1 8 10 9 5 9 9 4 4 6 9 89
53 Drenthe 9 3 2 1 5 6 6 2 8 8 2 4 5 9 70

Design of the HP4ALL study

54 Overijssel 9 1 1 1 2 5 7 4 8 9 1 1 3 9 61
55 Gelderland 10 2 1 2 2 5 6 4 5 7 4 3 4 6 61
56 Utrecht 10 1 2 3 1 4 5 4 6 7 3 3 4 6 59
57 Noord-Holland 10 1 3 2 2 4 6 7 7 9 6 1 2 8 68
58 Zuid-Holland 10 2 2 2 1 4 6 1 5 8 1 2 2 9 55
59 Zeeland 8 3 2 1 3 8 7 8 10 10 5 2 3 1 71
60 Noord-Brabant 10 1 3 2 1 3 3 6 3 3 7 7 8 2 59
61 Limburg 9 4 5 2 2 3 4 5 2 3 6 8 9 1 63
62 Flevoland 8 9 1 5 4 6 7 6 7 9 5 5 5 10 87

* ‘% PREG’: % Of pregnant women in the general population

* ‘AGE <20’: % Of teenage pregnancies
* ‘PRIMI’: % Of primiparous women
* ‘NW ETHN’: % Of pregnant women with a non-Western ethnicity
* ‘LOW SES’: % Of women in neighbourhoods with a socioeconomic status score <p20
* ‘ABS’: Absolute %
* ‘STND’: Standardised %
* ‘INEQ’: Inequality as measured by the relative risk of prevalences between women from neighbourhoods with soci-
oeconomic status score <p20 compared to >p80

115
Rotterdam; 3. Eindhoven; 4. Amsterdam; 5. Schiedam; 6. Almere ; 7. Delft; 8. Utrecht; 9. Maastricht;
10. Tilburg; 11. Heerlen; 12. Arnhem; 13. Friesland. According to the sum of the decile score for the
risk assessment sub-study (table 3) the following municipalities show the most adverse outcomes:
1. The Hague; 2. Amsterdam; 3. Rotterdam; 4. Arnhem; 5. Tilburg; 6. Nijmegen; 7. Schiedam; 8.
Utrecht; 9. Enschede; 10. Spijkenisse; 11. Heerlen; 12. Vlaardingen; 13. Groningen; 14. Leeuwarden10.
Additional to the identified municipalities, the province of Friesland is best qualified for the PCC
sub-study and the province of Groningen for the risk assessment sub-study.

Final selection municipalities

After the epidemiological selection of the candidate municipalities the list was first presented to
the Ministry of Health. The next step was to inform the Alderman and municipal health authorities
about their perinatal health status. They were invited to commit to the HP4All study. Criteria to
participate were: a) active involvement by a local Policy Officer (>one day per week for the duration
of the study), b) local political support for the study (e.g. financial support, involvement in health
related policy, local resources, involvement of local networks).
The following municipalities agreed to participate (see figure 3): in the province of Groningen
Appingedam/ Delfzijl/ Menterwolde/ Pekela and Groningen city, the municipalities of Enschede,
Nijmegen, Heerlen, Tilburg, Schiedam, Utrecht, The Hague, Amsterdam, and Almere. All
municipalities decided to participate in both sub-studies. As a separate municipal program on
reducing perinatal mortality was already being carried out in Rotterdam5, this city was not selected
for participation in the HP4All study. In these participating municipalities, general practitioners,
midwives, and obstetricians were approached for provision of the interventions.

INTRODUCTION TO THE SUB-STUDIES

The preconception care sub-study
This sub-study is a prospective cohort that aims to evaluate the effectiveness of individual
Preconception Care Consultations and the effectiveness of the employed recruitment strategy
for the PCC consultation services. Preconception care consultations are delivered by primary
caregivers (General Practitioners and midwifes) in the community. These consultations consist of
two sessions. Prior to the first session the woman fills in a questionnaire (www.zwangerwijzer.nl).
This questionnaire screens risk factors across the following domains: background, lifestyle, medical
history, obstetric/ gynecologic history, family, work/environmental. Thus, risk factor screening is
performed in a uniform way before the consultation. During the consultation a history is taken
regarding the presence of potential risk factors and an intervention plan is made with the women/
couple to reduce/ eliminate risk factors. Three months later a follow-up consultation is planned to
evaluate adherence to the intervention plan.
Uptake of individual PCC is known to be low. Thus additional efforts seem necessary to promote
uptake of the consultations.23 For this purpose a 4-tiered recruitment strategy is employed. Women
are informed about the PCC consultations by: (1) an invitational letter from the municipal health

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 H5 F3
FIGURE 3: Participating municipalities in the ‘Healthy Pregnancy 4 All’ project.

Design of the HP4ALL study

service or municipality, (2) invitational letter from the family doctor, (3) referral by the youth health
care service, (4) referral by a perinatal health peer educator.
The study population consists of women aged 18 – 41 years old. Participation is voluntary.
There are several primary outcomes. Firstly, the effectiveness of the PCC consultations in terms
of behavioral changes (use of folic acid supplements, smoking cessation, cessation of alcohol
consumption and illicit substances besides individual risk factors (e.g. obesity). Secondly, the
effectiveness of the recruitment strategy is assessed. We address this effectiveness by measuring
the extent to which each recruitment arm results in visitation of the PCC service and by the
characteristics of women that these recruitment strategies reach.
Women are enrolled in the cohort study after they have made an appointment for the PCC
consultation. When they participate they are asked to fill in a questionnaire and consent to
laboratory tests before each visit to the PCC health service. Biomarkers are tested to vouch self-
reported behavioral change of primary outcomes (erythrocyte folate,% carbohydrate transferrin
(CDT), serum cotinine levels and urinary drug tests). Furthermore anthropometric measurements
are collected at these two visits by the PCC provider. This data collection provides data for pre-
and post-measurements regarding PCC behaviors. Characteristics of women that visit the peer
education sessions are measured by questionnaires.

117
The antenatal risk assessment sub-study
In this cluster randomised trial (Trial registration: Dutch Trial Registry: NTR-3367) midwifery
practices in participating municipalities (‘clusters’) were randomly assigned to either the use of a
score card (‘R4U’) based antenatal risk assessment, care pathways and multidisciplinary consultation
(intervention group) or conventional risk assessment (control group).
The 70-item ‘R4U’ score card consists of six risk domains (social status, ethnicity, care, lifestyle,
medical history and obstetric history). Corresponding care pathways to both medical and non-
medical services will support health care professionals to encounter complex (non-)medical risk
factors. A predefined weighted sum risk threshold, based on weighted single risk factors, is derived
from the ‘R4U’ score card. If a pregnant woman’s individual sum risk score exceeds the threshold,
her case will be assessed in a multidisciplinary setting with community midwives, obstetricians,
and other care providers.
Score card based systematic risk assessment will be performed with the ‘R4U’ score card at the
first antenatal booking visit followed by (provided that informed consent is given), if necessary, a
specific referral to, e.g. a higher level obstetric care (gynaecologist), or psychosocial care in case
of medical or non-medical high risk using risk-specific care pathways. Additionally, these women
at increased risk will be reviewed in a multidisciplinary team of caregivers concerning tailored
antenatal care. We aim to assess 20% of all pregnant women in this multidisciplinary setting.
Participating midwives and obstetricians receive personal instructions in planned sessions by
the project team for the practical use of the web-based ‘R4U’ score card. Besides, an e-learning
program is available for all caregivers. The project team has developed 28 templates of care
pathways for all risk factors in the ‘R4U’ score card. Together with local healthcare professionals in
perinatal care, municipal services, community health services, and other services, these templates
will be adapted in organised meetings to local setting, taking the availability of local facilities,
agreements, and guidelines into consideration.
Pregnant women’s risk status in the control group is assessed conventionally, i.e. according to
the elaborate so-called ‘List of Obstetric Indications’ (in Dutch: Verloskundige Indicatie Lijst) which
lists all conventional (>140) high risk indications (for referral or consultation).26 In each control
municipality care ‘as usual’ will be provided until 700 participants have been included or until
2/3 of the study period (2 years) has passed. After that moment, the implementation of the risk
assessment intervention will start.
Primary outcomes are the prevalence of preterm birth and SGA, and the efficacy of ‘R4U’
implementation (measured by the number of ‘R4U’ score cards completed by the health care
professional against the number of booking visits, the development and use of care pathways
following ‘R4U’ scores, actual performed multidisciplinary consultations, and patient and healthcare
professional satisfaction).

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ORGANISATION AND TIME SCHEDULE
The study is rolled out by the national HP4ALL staff of the Erasmus Medical Center in Rotterdam and
by the local HP4ALL project managers. The staff consists of 2 junior researchers, research assistants
and 2 project managers (1 for each sub-study) and 2 program directors. The local project managers
are either allocated from the municipality or from the municipal health services. Organisation and
logistics regarding out roll of the two sub studies is presented in the specific design papers.
The HP4All study was initiated in April 2011. The HP4ALL research team was organised by May
2011. Municipalities had committed to participation in September 2011. Within the municipalities
local health care providers eligible to participation in the sub-studies were invited to participate as
of November 2011.

ETHICAL CONSIDERATIONS
The two HP4All sub-studies have been approved by the Institutional Board Review of the
Erasmus Medical Centre Rotterdam (Preconception Care sub-study: MEC 2012–425; Antenatal
risk assessment trial: MEC 2012–322). Participants in both studies will receive written and oral
5
information about the study after which informed consent will be obtained. Participation in either

Design of the HP4ALL study

sub-study is voluntary and no extra incentives will be provided. Health care providers participating
in both studies do not receive incentives. However in the PCC sub-study, providers will receive
reimbursement from the HP4All project, as PCC consultations are currently not covered by (most)
health care insurances.

DISCUSSION
In this study we described the set-up of the ‘Healthy Pregnancy 4 All’ study in which high perinatal
risk regions are targeted with two interventions based on preconception care and antenatal care.
The foundation of this study lies in the scientific and systematic analysis of the perinatal health
problem in the Netherlands. The study meets the current evidence to intervene early (before or
in pregnancy) upon risk factors associated with these perinatal health outcomes. By selection of
geographical areas, the study will intervene in potentially high risk populations that potentially will
benefit the most. We hypothesise that both strategies will contribute to the promotion of perinatal
health. In this project, optimal linkage is sought between curative and preventive care, public
health, government, and social welfare organisations. To our knowledge, this is the first study in
which these elements are combined.

CONFLICTS OF INTEREST
The authors declare that they have no conflicts of interest.

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AUTHORS’ CONTRIBUTIONS
All authors were involved in conception and design of the study. SD, JP, SVV, and AV drafted the
manuscript. All authors participated in the design of the study during several meetings. All authors
edited the manuscript and read and approved the final manuscript.

FUNDING
The research proposal was reviewed and funded by the Ministry of Health, Welfare and Sports of
the Dutch Government: grant 318804.

ACKNOWLEDGEMENTS
The Healthy Pregnancy 4 All study is made possible by financial support from the Dutch Ministry
of Health, Welfare and Sport, The Hague, grant 318804, and by financial support from participating
municipalities, health care insurer Menzis and ZonMw (The Netherlands Organisation for Health
Research and Development, grant number 50-52000-99).
Practical implementation of the study would not have been possible without all local project
coordinators: M. Bunnik, M. van der Ham, M. van der Biezen, G. Franssen, B. Zeelen, A. de Graaf, R.
Stoffels, G. de Rijk, B. Heusschen, S. Badal, E. Hochheimer, A. Verhoeff.
We would also like to thank all participating municipal health authorities, midwifery and GP
practices, obstetric outpatient clinics, research nurses, child welfare and social services, and the
perinatal health educators.
Special thanks goes to The Netherlands Perinatal Registry (kindly provided permission for data
analysis; number 11.36), The Health Authorities Netherlands, RIVM-Centre for Healthy Living, trial
coordinators and clinical chemists of all participating laboratories (Erasmus MC, ATAL-medial,
Atrium MC, SHO, Flevoziekenhuis, Certe, Labwest, MST, Saltro), the Erfocentrum, and the Advisory
Board of the Healthy Pregnancy 4 All study.
External advisors: J.M.W.M. Merkus MD PhD, [emeritus] professor of Obstetrics and Gynecology
and E.W. Steyerberg PhD, professor of medical decision making, Departments of Obstetrics and
Gynecology, and Public Health, Erasmus MC, Rotterdam, The Netherlands.
IT support: F. Santegoets and F. Buyuk Bozkoyun.

REFERENCES
1. Zeitlin J.R., Mohangoo A. European perinatal health report 2008: EURO-PERISTAT Project, with SCPE, EUROCAT, EURONEO-
STAT;2008.
2. Buitendijk SE, Nijhuis JG. [High perinatal mortality in the Netherlands compared to the rest of Europe]. Ned Tijdschr Ge-
neeskd. Sep 18 2004;148(38):1855-1860.
3. Mohangoo AD, Buitendijk SE, Hukkelhoven CW, et al. [Higher perinatal mortality in The Netherlands than in other Europe-
an countries: the Peristat-II study]. Ned Tijdschr Geneeskd. Dec 13 2008;152(50):2718-2727.
4. de Graaf JP, Ravelli AC, de Haan MA, Steegers EA, Bonsel GJ. Living in deprived urban districts increases perinatal health
inequalities. J Matern Fetal Neonatal Med. Mar 2013;26(5):473-481.

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5. Jaddoe VW, van Duijn CM, Franco OH, et al. The Generation R Study: design and cohort update 2012. Eur J Epidemiol. Sep
2012;27(9):739-756.
6. van Eijsden M, Vrijkotte TG, Gemke RJ, van der Wal MF. Cohort profile: the Amsterdam Born Children and their Develop-
ment (ABCD) study. Int J Epidemiol. Oct 2011;40(5):1176-1186.
7. de Graaf JP, Ravelli AC, Wildschut HI, et al. [Perinatal outcomes in the four largest cities and in deprived neighbourhoods in
The Netherlands]. Ned Tijdschr Geneeskd. Dec 13 2008;152(50):2734-2740.
8. de Graaf JP, Steegers EA, Bonsel GJ. Inequalities in perinatal and maternal health. Current opinion in obstetrics & gynecolo-
gy. Apr 2013;25(2):98-108.
9. Bonsel GJ, Birnie E, Denktas¸ S, Poeran J, EAP S. Dutch report: Lijnen in de Perinatale Sterfte, Signalementstudie Zwanger-
schap en Geboorte 2010 . Rotterdam: Erasmus MC; 2010. Available at: www.nvk.nl/Nieuws/Dossiers/Dossier Perinatalezorg.
aspx. Retrieved January 5, 2011.
10. van der Kooy J, Poeran J, de Graaf JP, et al. Planned home compared with planned hospital births in the Netherlands: intra-
partum and early neonatal death in low-risk pregnancies. Obstet Gynecol. Nov 2011;118(5):1037-1046.
11. Denktas S, Waelput A, van Voorst SF, et al. Healthy Pregnancy 4 All - Preconceptiezorg en risicoselectie tijdens de zwanger-
schap. Tijdschrift voor Gezondheidswetenschappen (TSG). 2012;90(8):479-483.
12. van Voorst SF, Vos AA, de Jong-Potjer LC, Waelput AJ, Steegers EA, Denktas S. Effectiveness of general preconception care
accompanied by a recruitment approach: protocol of a community-based cohort study (the Healthy Pregnancy 4 All study).
BMJ Open. 2015;5(3):e006284.
13. Vos AA, van Voorst SF, Waelput AJ, et al. Effectiveness of score card-based antenatal risk selection, care pathways, and
5
multidisciplinary consultation in the Healthy Pregnancy 4 All study (HP4ALL): study protocol for a cluster randomized con-

Design of the HP4ALL study

trolled trial. Trials. 2015;16:8.
14. Barker DJ, Bagby SP, Hanson MA. Mechanisms of disease: in utero programming in the pathogenesis of hypertension. Na-
ture clinical practice. Nephrology. Dec 2006;2(12):700-707.
15. Mook-Kanamori DO, Steegers EA, Eilers PH, Raat H, Hofman A, Jaddoe VW. Risk factors and outcomes associated with
first-trimester fetal growth restriction. JAMA. Feb 10 2010;303(6):527-534.
16. Saravelos SH, Regan L. The importance of preconception counseling and early pregnancy monitoring. Seminars in repro-
ductive medicine. Nov 2011;29(6):557-568.
17. Korenbrot CC, Steinberg A, Bender C, Newberry S. Preconception care: a systematic review. Matern Child Health J. Jun
2002;6(2):75-88.
18. van der Zee B, de Beaufort I, Temel S, de Wert G, Denktas S, Steegers E. Preconception care: an essential preventive strategy
to improve children’s and women’s health. J Public Health Policy. Aug 2011;32(3):367-379.
19. the Health Council of the Netherlands. Preconception care: a good beginning [Preconceptiezorg, een goed begin]. the
Hague, the Netherlands2007.
20. Amelink-Verburg MP, Buitendijk SE. Pregnancy and labour in the Dutch maternity care system: what is normal? The role
division between midwives and obstetricians. Journal of midwifery & women’s health. May-Jun 2010;55(3):216-225.
21. de Graaf JP, Ravelli AC, Wildschut HI, et al. [Perinatal outcomes in the four largest cities and in deprived neighbourhoods in
The Netherlands]; Perinatale uitkomsten in de vier grote steden en de prachtwijken in Nederland. Ned Tijdschr Geneeskd.
Dec 13 2008;152(50):2734-2740.
22. Denktas S, Bonsel GJ, Van der Weg EJ, et al. An Urban Perinatal Health Programme of Strategies to Improve Perinatal Health.
Matern Child Health J. Aug 26 2011.
23. Poeran J, Maas AF, Birnie E, Denktas S, Steegers EA, Bonsel GJ. Social deprivation and adverse perinatal outcomes among
Western and non-Western pregnant women in a Dutch urban population. Soc Sci Med. Apr 2013;83:42-49.
24. [Perinatal care in the Netherlands 2008]. The Netherlands Perinatal Registry. Utrecht2008.
25. Roalfe AK, Holder RL, Wilson S. Standardisation of rates using logistic regression: a comparison with the direct method.
BMC health services research. 2008;8:275.
26. Health Insurance College Verloskundig Vademecum, Health Insurance College, Diemen; 2003.

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EFFECTIVENESS OF GENERAL
PRECONCEPTION CARE
ACCOMPANIED BY A RECRUITMENT
APPROACH: PROTOCOL OF A
COMMUNITY-BASED COHORT STUDY
(THE HEALTHY PREGNANCY 4 ALL STUDY)
S.F. van Voorst, A.A. Vos, L.C. de Jong-Potjer, A.J.M.
Waelput, E.A.P. Steegers, S. Denktaş
BMJ Open 2015; 5: e006284
ABSTRACT
Introduction: Promotion of healthy pregnancies has gained high priority in the Netherlands
because of the relative unfavourable perinatal outcomes. In response, a nationwide study
Healthy Pregnancy 4 All (HP4All) has been initiated. One of the substudies within HP4All focuses
on preconception care (PCC). PCC is an opportunity to detect and eliminate risk factors before
conception to optimise health before organogenesis and placentation. The main objectives of
the PCC substudy are (1) to assess the effectiveness of a recruitment strategy for the PCC health
services and (2) to assess the effectiveness of individual PCC consultations.
Methods/analysis: Prospective cohort study in neighbourhoods of 14 municipalities with perinatal
mortality and morbidity rates exceeding the nation’s average. The theoretical framework of the
PCC substudy is based on Andersen’s model of healthcare utilisation (a model that evaluates
the utilisation of healthcare services from a sociological perspective). Women aged 18 up to and
including 41 years are targeted for utilisation of the PCC health service by a four armed recruitment
strategy. The PCC health service consists of an individual PCC consultation consisting of (1) initial
risk assessment and risk management and (2) a follow-up consultation to assess adherence
to the management plan. Primary outcomes regarding the effectiveness of consultations is
behavioural change regarding folic acid supplementation, smoking cessation, cessation of alcohol
consumption and illicit substance use. The primary outcome regarding the effectiveness of the
recruitment strategy is the number of women successfully recruited and the outreach in terms of
which population is reached in comparison to the approached population. Data collection consists
of registration in the database of women that enrol for a visit to the individual PCC consultations
(women successfully recruited), and preconsultation and postconsultation measurements among
the included study population (by questionnaires, anthropometric measurements and biomarkers).
Sample size calculation resulted in a sample size of n=839 women. 

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BACKGROUND
The Healthy Pregnancy 4 All (HP4All) study was initiated because of the relatively high perinatal
mortality rate of 10 per 1000 births, ranking the Netherlands at an unfavourable position in
Europe.1 A huge concern was the inequality in the perinatal mortality rate within the country:
deprived neighbourhoods were found to have an up to four times higher perinatal mortality rate
than average.2 Societal, professional, and political debates about how to improve perinatal health
in the Netherlands dominated the policy agenda. One of the results was the launch of the HP4All
study - commissioned by the Dutch Ministry of Health and Welfare, in May 2011.3,4
The objective of the HP4All study is to develop evidence based strategies to improve perinatal
health by interventions in the preconception or the antenatal period, which reduce adverse
pregnancy outcomes. Accordingly, HP4All is divided into two sub-studies: the Preconception Care
study (PCC) - a prospective cohort study - and the antenatal Rotterdam Risk Assessment (R4U)
study – a cluster randomized controlled trial.5 This paper concentrates on the PCC sub-study.

The rationale of PCC and its delivery approaches

The rationale of preconception care (PCC) originates in the growing recognition that the embryonic
development and placentation phase is critical for the outcome of the pregnancy.6 PCC is a set of 6
interventions that aims to identify and modify biomedical, behavioral, and social risks to a woman’s
health or pregnancy outcome through prevention and management.7

The HP4ALL preconception care sub-study

The preconception period can be seen as the earliest link between maternal and newborn
health. Therefore it has been recognized as a pivot point which can be utilized to improve perinatal
health.7,8,9 In the Netherlands, 82% of the perinatal mortality cases were preceded by small for
gestational age (SGA), premature birth and congenital anomalies.10 In theory many risk factors
for these problems are present and potentially detectable and treatable/ manageable before
conception.6,8 By the time a women enters antenatal care a large part of organogenesis has taken
place. PCC should therefore be regarded as a necessary additional component to the obstetric care
system, in the improvement of perinatal outcomes.

Organization of PCC
PCC can be organized in three forms: (1) collective PCC consisting of interventions targeted at
the general public (e.g. with group education or national campaigns); (2) general individual PCC
consisting of individual consultations among couples contemplating pregnancy amongst the
general public; (3) specialist individual PCC consisting of individual consultations amongst couples
contemplating pregnancy with complex risk factors.11 These forms can be integrated in different
approaches for delivery, dependent on the health system (e.g. primary care, hospital based, PCC
clinic) and the targeted audience (e.g high-risk population or general public).12

125
 Individual PCC is a unique opportunity for professionally led PCC – which addresses both
general risk factors and personal risk factors after systematic screening. Furthermore, individual PCC
is an opportunity to deliver PCC in a responsive fashion – so that besides the systematic standard
risk factor screening the individual patient’s needs or preferences can be met.13 Thus, individual PCC
consultations are seen as the form of PCC to implement in the PCC sub-study.

Recruitment for PCC

The concept of visiting a health care professional for PCC is not common in the Netherlands as
well as in many other countries. Firstly, the uptake of PCC requires that a pregnancy is planned. An
explanation for low uptake of PCC can be sought in the unfamiliarity of women or couples with the
availability and the potential benefits of the health service.14-16 Women or couples could assume
that they are healthy and that it is not necessary to discuss their pregnancy wish with a professional.
Women who are aware of risks might believe that nothing can be done to influence the course of
a pregnancy in the future. Besides beliefs, actual barriers to attend PCC could also play a role. For
instance a woman’s or couples willingness to disclose their pregnancy wish to a professional is a
known barrier.16 Simply delivering PCC to women or couples upon request does not seem to be
sufficient to provide PCC at a scale to improve perinatal health. An active recruitment approach
is necessary. Different active recruitment approaches are described in the literature. Wallace et
al., describe that the opportunistic approach of women during daily care is utilised most often by
primary caregivers.17 However, despite the fact that couples in the general public are known to
have at least more than one risk factor,18 many women/couples of reproductive age do not request
a PCC consultation from their general practitioner (GP). An opportunistic approach is not feasible
for midwives as they rarely see non-pregnant women. One trial employs the approach of inviting
women through invitational letters.19 These approaches, however, require efforts from caregivers
in an already stressed system; and the effectiveness is unknown. An evidence-based strategy
is necessary to create an outreach for individual PCC consultations in order to improve perinatal
health on a larger scale.
The main objective of this study is to implement and evaluate a local recruitment strategy
for individual PCC and to promote and evaluate (health) behaviour change by delivery of PCC
consultations in primary care. This paper provides an overview of the design and the methodology
of the HP4All PCC study.

METHODS/ DESIGN
Key attributes of the study:
The PCC study is designed as a community-based study with a high-risk approach in a primary care
setting with tools to improve the uniformity of the health message.
This section describes the key concepts of the study:
• The high-risk approach: The Dutch Ministry of Health, Welfare and Sport, commissioned the
HP4All study to target high-risk populations. After ranking the municipalities with the highest

126
 perinatal mortality and morbidity in the country, 14 municipalities were selected as candidates
for participation (selection described elsewhere).4 The key approach of HP4All is to roll out the
interventions in high-risk neighborhoods – meaning neighborhoods with rates of adverse
perinatal outcomes above the average of the selected municipalities. It is presumed that
women in these neighborhoods can benefit the most from interventions in the HP4All study.
Prevalence of risky lifestyle behaviors in the preconception phase is not exactly known as there
is no surveillance amongst women contemplating pregnancy and risky behaviors tend to be
underreported during pregnancy. With regards to folic acid supplementation, a recent Dutch
study conducted amongst a multiethnic population reported that 40% of pregnant women
to have used folic acid supplements before conception.20 A different study reported that of
7106 pregnant women, 8% smoked in the first trimester only and 17% continued smoking
throughout pregnancy.21 In total, 35% - 50% of women are estimated to continue their alcohol
consumption during pregnancy.22 The HP4ALL study will provide more information regarding
risk behaviors before and in pregnancy in these high-risk neighborhoods.
• The community-based approach: A community-based approach has advantages as it (1)
reaches populations which are hard to reach by ensuring trust: collaboration with local health
authorities in a community-based approach provides the opportunity that the target population 6
is approached by professionals they know and mostly trust; (2) promotes collaboration and
local support as it draws the sectors together necessary for optimal outreach.23

The HP4ALL preconception care sub-study

• A primary care approach: GP’s and midwives practicing in the high-risk neighborhoods are
recruited to deliver individual PCC consultations. They were selected because they can deliver
the intervention in a responsive fashion, because they are familiar with the target population
and by arranging them to provide the PCC consultations, accessibility of PCC is ensured.
• Tools for the consistency of health messages: PCC for the general population necessitates
a thorough risk factor screening. Therefore GP’s and midwives are facilitated with tools and
training to ensure consistency, which is important as different health messages reduce the
effectiveness of interventions.13

Theoretical framework of the HP4All PCC sub-study

The delivery of PCC in the PCC sub-study is based on the Andersen’s model of healthcare
Utilization.24 Andersen defines the utilization of a health service and other personal health
behaviors as an outcome. These outcomes are a function of the predisposition (to utilize the health
care service) and enabling or impeding factors (to utilize the health care service) and perceived and
objective need (to utilize the health care service). These factors originate in the individual which
interacts with his/her context. This model has been used to understand utilization of PCC services
and other health care services (oral health services, mental health services, primary care).25,26
Figure 1 should be read from left to right: The PCC sub-study intervenes in the environment in order
to target the population to achieve the outcomes on the right.
• The environment: The study entails two organizational changes in the environment: Firstly,
individual PCC consultations are made available. Secondly, a strategy to recruit eligible women
to utilize the PCC services is employed (see Figure 2).

127
 H6 F1
Framework of the Healthy Pregnancy 4 All - Preconception Care Sub-Study

Environment Interaction with the targeted population Outcomes

Interventions in local Predisposing Utilization of PCC health

Enabling Resources Need
community: Characteristics : services

1. PCC consultations Demographic factors:
Age, Marital status, Ethnicitiy,
parity, having a child, pregnancy
intention
2. Recruitment strategy
In the community:
Barriers to utilize care Perceived need:
Perceived health, fertility
concerns, obstetric history
Individual factors:
Education, income, information
skill, time Objective need:

128
Social influences: Chronic illness or disease ,
Social contacts , religion, subfertility, obstetric history,
structural and social acculturation preconception risk factors Behavioural change
External Environment
regarding PCC risk factors

Health beliefs, attitudes and

- Organization of local primary care network knowledge:
- Compeding projects in Fetomaternal health Towards a ) preventive health
- Professional organizations care b) caregivers c) PCC
- Political priorities in local health care d) pregnancy
- Position of primary care in the health system
- Insurance companies
FIGURE 1: The framework for the Healthy Pregnancy 4 All – Preconception care substudy
• The population: The study aims to target women of reproductive age (defined as 18 up to and
including 41 years) living in municipalities with high perinatal mortality and morbidity rates
with a specified recruitment strategy. Women will however decide individually, within their own
context, whether they will use the health care service and/or change their health behaviours.
We hypothesize this decision to be a function of:
·· Predisposing factors: In accordance with Andersen’s model of health care utilization we
contemplate that a woman’s decision to utilize PCC will depend on a function of her health
beliefs and attitude towards the preconception health service and a healthy pregnancy,
social influences and demographic factors.
·· Enabling factors: The targeted woman can be stimulated to visit (or impeded from visiting)
the PCC health service by community factors (e.g. a good infrastructure to attend a PCC
consultation) or by individual factors (e.g. speaking a different language than the PCC care
provider can be a barrier to attending the PCC consultations).
·· Need: The targeted woman needs to feel a need to utilize the service. There are two kinds
of need: (1) an objective need (the service is necessary in terms of medical risks) and (2)
a perceived need (the service is perceived as necessary by the woman herself ). Perceived 6
need can be related to the objective need, but this is not necessarily the case. Factors
from the literature or those of which we hypothesized to influence the objective and/

The HP4ALL preconception care sub-study

or perceived need for a PCC consultation are mentioned in Figure 1. Need, as a resultant
of a perceived need and objective need is thought to be influenced by predisposing
characteristics (e.g. knowledge regarding risks, social network to point out the relevance of
PCC for the individual).
• The outcomes: The primary outcomes of the PCC sub-study are reduction of preconception risks
by (1) the utilization of PCC health services and (2) behavioural change regarding preconception
risk behaviours. Reduction of preconception risk factors is thought to subsequently reduce (the
risk for) perinatal morbidity and mortality.

FIGURE 2: The recruitment strategy of the Healthy Pregnancy 4 All—Preconception Care substudy

Child Peer
Welfare Health Midwives
Service Educators

General
Practitioner
TARGET
POPULATION
Municipality

129
The intervention
As displayed in Figure 3, the Preconception Care Consultation consists of 2 visits to a participating
General Practitioner or midwife in the course of 3 months. During the first consultation systematic
risk assessment is performed and a tailored management plan to address risk factors. Caregivers
evaluate whether goals are reached or whether additional measures are necessary. Consultations
are supported and archived with PCC tools (see Box 1).27,28 The tool for risk assessment
(ZwangerWijzer in Dutch) is a validated tool.29 When women participate in the cohort study as
study participants extra data are collected to assess the effectiveness of the provided care (see
section on data collection).

Study population
All women aged 18 up to and including 41 years (the adult reproductive life span) who make
an appointment at the PCC health service are enrolled (registered in the database) to assess the
effectiveness of the recruitment strategy. The additional criteria for inclusion in the cohort study

FIGURE 3: Flowchart for enrollment and intervention and data collection

HP4ALL recruitment strategy

Data collection:
Enrollment for a PCC - registry of consultation
consultation - demographic characteristics
- method of recruitment

Counselling for participation in

the effectiveness trial

Exclusion Inclusion

Data collection:
First consultation:
- questionnaire 1
Risk assessment and formulation
- anthropometric measurements 1
of an individual plan
- laboratory samples
: blood and urine

3 months

Data collection:
Second consultation:
- questionnaire 2
Evaluation of adherence to the
- anthropometric measurements 2
management plan
- laboratory samples
: blood and urine

130
to assess the effectiveness of PCC are: (1) a pregnancy wish (regardless of in which phase) and
(2) voluntary participation. Exclusion criteria are: (1) no permission to be encountered about
participation in the study (2) non-response to approach for inclusion (3) not speaking one of the
following languages (Dutch, English, Turkish, Polish or Arabic) (4) cancellation/ no-show at the
appointment.

Recruitment and enrollment

The recruitment strategy consists of four components (see Figure 2):
(1) an invitational letter from the municipal public health service;
(2) an invitational letter from participating General Practices to their own patients;
(3) Youth Health Care Physicians or nurses inform parents who visit the routine check-ups at the
youth health care center for their child with an information leaflet containing the names of the
participating practices;
(4) Referral by a preconception health educator after PCC education sessions. (A preconception
health educator is a person from the peer group (the local community) who has completed a
certified training in health communication skills/ preconception health). 6
All female applicants for the PCC health care services are registered. These women are sent an
information letter followed by a telephone call for individual counselling about participation in the

The HP4ALL preconception care sub-study

study by the research team. Participants who agreed to take part in the study signed an informed
consent form. Women receive the same PCC regardless of participation in the study; participation
requires the participant to partake in data collection (questionnaires and laboratory tests) parallel
to the care she receives.

Outcomes
The primary outcome regarding the effectiveness of the individual PCC consultations is
behavioral change. Behavioral change is assessed for folic acid supplementation, smoking, alcohol
consumption and illicit substance use. These four preconception health behaviors were chosen
as primary outcomes due to their prevalence, their impact on the fetus and modifiability.30-35
Differences in these behaviors are assessed by premeasurement and post-measurement by
questionnaires (self-reported changes) and biomarkers (biochemical assessment of behavioral
change). Biomarkers are used, as it is known that self-reported outcomes can show socially
desirable answers. For example in case of the use of folic acid supplements a Dutch study found an
over-report of 22% for self-reported folic acid supplement use.20
The primary outcome regarding the effectiveness of the recruitment strategy is the utilization
of the preconception care services of the program. This is measured quantitatively by the number
of women that utilized the preconception care program (women successfully recruited) in relation
to the number of women approached by the recruitment strategy. Secondly, the effectiveness of
the recruitment strategy is assessed in terms of outreach: by assessing characteristics of women
reached. This includes basic demographics of women that were successfully recruited and

131
identification of predisposing factors, need, and enabling factors according to Andersen’s’ model
of health care utilization amongst women included in study. Data regarding the target group (all
women aged 18 up to and including 41 years in the geographically targeted area) is obtained from
municipal administrative records.
Outcomes, measures and data sources are presented in Table 1.

Data collection and measurement

BOX 1: Tools for delivery of PCC in the Healthy Pregnancy 4 All - PCC sub-study

Standardised risk assessment instruments improve delivery of PCC; unify risk assessment and facilitate documentation
needs. The Healthy Pregnancy 4 All programme uses two tools:

The ZwangerWijzer Tool:

ZwangerWijzer is a self-administered questionnaire for couples designed to be filled in prior to consultation.
The questionnaire is freely accessible on the internet or on paper.28 The webbased survey has additional features: (1)
additional information is provided when a risk factor is present (2) a list of risk factors is generated; listing what should be
discussed during the PCC consultation. This list can be emailed to the PCC caregiver.
The questionnaire is adopted from The Preconception Health Assessment Form47 and a Family History Survey. It covers
the following risk domains and risk factors:

Background Lifestyle Medical history Obstetric and Family Work

Gynecologic his-
tory

Maternal age Exposure to radi- STD’s Prior pregnancies Family history Chemical expo-
BMI ation HIV Pregnancy com- consanguinity sure
Ethnicity Smoking Rubella vaccina- plications Other exposure
Alcohol tion Uterine anom- Infectious agents
Drugs (Chronic) illness alies Shifts/ irregular
Eating disorders Prescribed medi- Prior gynecologic hours
Nutrition cation surgeries Physically de-
Folic acid supple- Over-the-counter manding work
ment use drugs Stress
Vitamin A
Toxoplasmosis
Listeria

The Preconceptiewijzer Tool:

Preconceptiewijzer is a web based PCC archive system complementary to the ZwangerWijzer questionnaire.27 Provid-
ers can create a PCC file for their patients in which the questionnaire can be archived and the consultation(s) can be
documented. The tool provides an overview sheet in which present risk factors (identified in ZwangerWijzer) are linked
to digital patient information leaflets and protocols for the caregiver about these risk factors. The latter is a measure to
improve the uniformity of health messages and interventions. Preconceptiewijzer is available online; providers have an
own account which is secured for own use. This account and technical support is freely available.

132
The process of data collection is illustrated in Figure 3.
All women who make an appointment are registered in the Gemstracker (Generic Medical
Survey Tracking system) database.36 The Gemstracker system firstly helps keep a log of all
consultations. Furthermore, the system assists in keeping a log of the inclusion process after which
it organizes data collection within a specific time track, by activating fields or questionnaires for
respondents or PCC providers.
Data collection consists of a questionnaire (questionnaire 1 and 2 respectively), anthropometric
measurements and laboratory tests, performed as a baseline measurement (before the first
consultation) and follow-up measurement (around the second consultation).
• Questionnaires: Questionnaire 1 is filled in prior to the first PCC visit. This questionnaire
assesses the characteristics of the study participant and health behaviors regarding the primary
outcomes and other preconception risk factors. Questionnaire 2 is filled in after three months
to assess changes in health behavior regarding preconception risk factors. Questionnaires
are filled in on paper or via internet. Participants were reminded up to two times to fill in the
questionnaire. The questionnaires were available in Dutch, English, Arabic, Turkish and Polish.
• Anthropometric measurements: PCC providers measure the following anthropometric
measurements at both PCC visits: blood pressure (mmHg), length (cm’s), weight (kg), hip and 6
waist circumference (cm). These measurements were performed according to a predefined
protocol.

The HP4ALL preconception care sub-study

• Laboratory tests: Data from the questionnaires regarding the primary outcomes of behavioral
change are verified with biomarkers. Folic acid supplementation is assessed by measuring red
cell folate in the serum.32 Smoking cessation is assessed by serum cotinine levels.37,38 Applying
biomarkers for alcohol consumption is challenging because diagnostic accuracy is generally
moderate and diagnostic properties differ over different alcohol consumption quantities
and patterns. Ideally one would match a biomarker to the presumed alcohol consumption
(quantitatively in time) of the study population. However, the prevalence of alcohol
consumption is difficult to predict as there is a lack of consensus regarding the classification of
alcohol consumption.39 Numbers in the general population and in cohorts of pregnant women
vary.34 As there is no consensus regarding safety of alcohol consumption in the preconception
phase the recommendation is to not consume alcohol in the preconception phase. Thus, we will
be interested in biomarkers for any level of alcohol consumption. To detect alcohol drinking in
the heavy end of the spectrum we chose to use Carbohydrate deficient transferrin (CDT%).40 As
a biomarker to assess the mild to moderate drinking spectrum we will explore the availability
of ethylgluconeride (EtG) or a serum test of phosphatidylethanol (PEth) for the mild-moderate
drinking spectrum.41 Illicit substance use will be tested with conventional urinary drug tests
(assessing amphetamines, barbiturates, benzodiazepines, cannabinoids, cocaine, methadone,
opioids (codeine, morphine, heroin, oxycodone etc.), phencyclidine, propoxyphene, synthetic
cannabinoids). Providers are blinded to data from questionnaires and from the mentioned
biomarkers.

133
In case of no-show at either consultation, PCC providers are encouraged to provide a new
appointment for the consultation. If the woman does not attend the first PCC consultation she will
be included for the analysis of the recruitment strategy. However, she will be excluded from the
PCC cohort. If a woman does not attend the second PCC consultation she will be asked to complete
her second questionnaire and where it is logistically opportune she will be asked to undergo a
second laboratory assessment. She will be included in the outcome assessment of the effectiveness
trial in that case.

Data analysis
Characteristics of the study population and preconception health behaviors at baseline will be
described continuously (mean or median, SD or IQR range) or descriptively (percentages and CI’s).
Changes in preconception health behaviors will be analyzed paired. Preconsultation and post
consultation differences will be expressed with mean differences and SD’s in continuous variables
(in case of normality) or with median and IQR (in case of skewed data). Students T tests (in case of
normality) or Wilcoxon Matched Pairs Test (in case of skewed data) will be performed for hypothesis
testing. Dichotomous variables will be tested with the exact binomial test. Significance testing
will be performed two sided with an α of 0.05. Regarding change in folic acid supplementation
and smoking cessation one-sided testing will be performed with an α of 0.025 (in line with the
hypothesis used for sample size calculation).

TABLE 1: Outcome assessment listed per study aim.

RESEARCH AIM Outcome measure Data source

EFFECTIVENESS OF INDIVIDUAL PCC CONSULTATIONS

Primary Folic acid suppletion Self-reported folic acid use. Questionnaire and blood analysis at first
Outcomes Biomarker (erythrocyte folate) confirmed consultation and 3 months after first
folic acid suppletion. consultation.

Smoking Self-reported smoking cessation. Questionnaire and blood analysis at first

Biomarker (serum cotinine) confirmed consultation and 3 months after first
smoking cessation. consultation.
Alcohol Self-reported cessation of alcohol con- Questionnaire and blood / urine analysis
sumption. at first consultation and 3 months after
Self-reported reduction of alcohol first consultation.
consumption.
Biomarker (serum %CDT; urinary EtG or
PeTH) confirmed reduction or cessation
of alcohol consumption.
Illicit substance use Self-reported cessation of illicit substance Questionnaire and urine analysis at first
use. consultation and 3 months after first
Biomarker (drug assessment in urine) consultation.
confirmed cessation of illicit substance
use.

134
TABLE 1 continued
RESEARCH AIM Outcome measure
EFFECTIVENESS OF RECRUITMENT STRATEGY
Primary Characteristics of the Characteristics of women who utilized
Outcomes cohort measured by the PCC health service according to the
Andersen’s’ model framework of the sub-study (figure 1).
Outreach of the Proportion of women successfully
municipal letter recruited through the letter from the
municipality in relation to the number
of women approached by the municipal
health service/ municipality.
Characteristics of women successfully
recruited after receiving the letter from
the municipality in relation to characteris- target population: all women aged 18-42
tics of women residing in the selected
neighborhood(s).
Outreach of the GP Proportion of women successfully recruit- Data on women successfully recruited
letter ed in relation to the number of women
approached by a letter from their general
practice.
Characteristics of these women in rela-
tion to characteristics of women residing
in the selected neighborhood(s).
Outreach of the Pre- Proportion of women successfully
conception health recruited for individual PCC consultation
educators after being approached about the service women included in the study (question-
during a peer health education session.
Characteristics of these women in rela-
tion to characteristics of women residing
in the selected neighborhood(s).
Outreach of the Proportion of women successfully re-
Child welfare service cruited after being approached about the (Gemstracker database) and data from
service during a visit at the Child Welfare
services.
Characteristics of these women in rela-
tion to characteristics of women residing
in the selected neighborhood(s).
135
Data source
Questionnaire at first consultation.
Data on women successfully recruited
(the Gemstracker database) and data
from women included in the study
(questionnaire 1).
(Anonymous) municipal administrative
records provide characteristics of the
residing in the high risk neighborhood.
6
(the Gemstracker database) and data
from women included in the study
(questionnaire 1).
(Anonymous) register of women who
The HP4ALL preconception care sub-study
were sent a letter by general practices.
(Anonymous) municipal administrative
records provide characteristics of the
target population: all women aged 18-42
residing in the high risk neighborhood.
Data on women successfully recruited
(Gemstracker database) and data from
naire 1).
Questionnaires of participants of precon-
ception health education sessions.
(Anonymous) municipal administrative
records provide characteristics of the
target population: all women aged 18-42
residing in the high risk neighborhood.
Data on women successfully recruited
women included in the study (question-
naire 1).
(Anonymous) municipal administrative
records provide characteristics of the
target population: all women aged 18-42
residing in the high risk neighborhood.
 Regarding the effectiveness of the recruitment strategy utilization of the PCC health service will
be expressed in percentages in relation to the number of women approached by the recruitment
strategy. Characteristics of women who visit the PCC health service will be assessed according to
the framework for utilization of the PCC service (see Figure 1).

Sample size calculation

Sample size calculation was performed for the two most important primary outcomes: folic acid
supplementation and smoking cessation. Regarding folic acid supplementation, 839 women are
needed in order to reject the null hypothesis (H0) that the PCC service will lead to a 20% increase
of folic acid users in women who were not already using folic acid supplements at baseline
(assumptions for this power calculation were (1) the smallest clinically relevant difference (‘Δ’) is
a 20% increase of folic acid in non-users at baseline, (2) the proportion of women using folic acid
at baseline is 30% (π0=30%)42, (3) a select drop-out rate of 10%, (4) pairwise analysis of results, (5)
a statistical significance level of α<0.025 (one-sided correction for multiple testing due to primary
outcome measures) and (6) a power (1-β) of 0,80). Regarding smoking cessation, 687 women are
needed to reject the null hypothesis (Ho) that the PCC service will lead to a <5% decrease of smoking
cessation, amongst women who smoked at baseline. (Assumptions for the power calculation were
(1) the smallest clinically relevant difference (‘Δ’) is a 5% decrease in smoking compared to baseline,
(2) the proportion of women smoking at baseline is 30% (π0=30%)43,44 (3) the a- select drop-out
rate of 10%, (4) pairwise analysis of results, (5) a statistical significance level of α<0.025 (one-sided
correction for multiple testing due to primary outcome measures) and (6) a power (1-β) of 0.80.)
Thus, the cohort study should comprise 839 women to meet the needs of both primary outcomes.

Organization and time schedule

Municipalities were encountered for participation from June - November 2011. General Practices
and midwife practices were encountered for participation from November 2011 to July 2013.
Practices were prepared to deliver PCC in the PCC sub-study after a one-on-one training to
deliver PCC according to the study protocol. They received a self-study e-learning course and study
material about preconception care in general and about risk factors. Practices were provided with
information leaflets, posters and kits to hand out for the laboratory tests. Recruitment strategies
were rolled out when practices were ready to receive participants for the study. Data collection is
performed in close collaboration with the practices by means of the Gemstracker system. In order
to promote the readiness of study participants to provide blood and urine samples and to ensure
timely handling of the samples, all laboratory sampling and processing is done at neighborhood
health centers or local laboratories in the participating municipalities. Finally, to reduce bias, the
non-time critical laboratory tests are performed at one central laboratory (the trial laboratory of the
Erasmus Medical Center of Rotterdam, the Netherlands).
After each PCC consultation study participants receive a preassembled laboratory kit form
their PCC provider. This kit includes 1mL freezer capsules, a urine container and an application
form to process the material according to the standard operating procedure of our study. All local

136
laboratories were accredited by CCKL (in Dutch: Coördinatie Commissie ter bevordering van de
Kwaliteitsbeheersing op het gebied van Laboratoriumonderzoek in de Gezondheidszorg or the
organization that audits laboratories in the health care system in the Netherlands).45 Blood samples
are collected in EDTA, SST or sodium fluoride vacutainers (in size and numbers as routine to the
local laboratories). Local laboratories perform tests which have to be performed within 1 hour (e.g.
glucose) or before refrigeration (hemoglobin, hematocrit, mean corpuscular volume, red blood cell
count) directly after blood sampling. Two ml of whole blood is then pipetted and stored and the
remainder is centrifuged (depending in local equipment: ±2000g/ 10 minutes). Approximately 2ml
of plasma and 7ml of serum is pipetted into 1ml freezer capsules. Urine is centrifuged (depending
in local equipment: ±2000g/ 10 minutes) after which 4 ml is stored. Whole blood is stored at -20°C;
plasma/serum fraction and urine at -70 or 80°C (depending on local equipment). All laboratories
closely monitor their storing protocol and are able to provide a report of the storing conditions at
request. At set times all samples are distributed to the central trial laboratory.
The first municipality started enrollment in February 2013 and the last municipality started
inclusion in February 2014. Enrollment is ongoing until time period: April 2014 or until the
calculated sample size of included participants has been reached to meet current research goals.
As the sample size has not been reached as yet, the inclusion period is currently planned until 6
December 2014.

The HP4ALL preconception care sub-study

Ethical considerations
The HP4All PCC sub-study has been approved by the Medical Ethical Committee of the Erasmus
Medical Center of Rotterdam (MEC 2012-425). In line with regulations an independent physician is
available for consultation by the (eligible) study population.

DISCUSSION
The Healthy Pregnancy 4 All – Preconception Care sub-study aims to provide evidence for
comprehensive and systematic delivery of PCC to the general public that contemplates pregnancy
and to identify effective ways to reach women to promote utilization of the PCC health services.
The study is rolled out in municipalities with disadvantaged perinatal health. Outcome measures
of the study are the effectiveness of the employed recruitment strategy and the effectiveness of
the PCC service in achieving behavioral change regarding preconception risk behaviors. In doing
so, we acknowledge that merely providing PCC is insufficient and we aim to develop an integrated
approach in which recruitment is combined with delivery of PCC – ultimately to improve perinatal
health care by PCC.
Internationally PCC is implemented in various ways and within different settings. This study will
place PCC in a cross-domain perspective in the Netherlands for the first time as multidisciplinary
collaborations are initiated amongst municipalities, public health services and the curative care
setting. Although this can be a strength, it might also be challenging to motivate different partners
with different points of departure in the health system. Prior to this program for instance the role

137
of public health in fetal-maternal health care was very limited. Furthermore, PCC has been brought
under attention within general practice and midwifery. However the experience seems to be that
PCC is at best, only delivered at a small scale within these echelons. By implementing PCC in these
echelons in the context of the current study, more can be learnt about what is necessary to upscale
delivery of PCC, if effective.
A limitation in our approach to improve perinatal health outcome with PCC is that we target the
group that plans pregnancy, as this is a precondition for PCC. Although the Netherlands has a high
planned pregnancy rate and excellent access to contraceptives,11,46 planned pregnancy rates and
the access to contraceptive care could be lower within a population with lower socio-economic
status. Initially, we aim to optimize outcomes of planned pregnancies. If this is effective, it would
be the time to assess where PCC and family planning could be integrated to increase further
effectiveness.
A limitation in the assessment of the effectiveness of PCC consultations will be that the
sampled population could be prone to a participation bias. First, since the eligible population will
rely on the extent to which the recruitment strategy is able to recruit a study population that is
representative of the community. Second, because eligible high-risk women might be more difficult
to include in the effectiveness study. However, we believe that we will have the data to explore the
representativeness of the included population in relation to the population that did not want to
participate in the cohort study and in relation to the targeted population in the community.
This is one of the first cohort studies in the Netherlands that assesses effectiveness of a PCC
approach in a high-risk area in a general practitioner and midwifery setting. We have future
aspirations to do further research within the context of the current study.

AUTHORS’ CONTRIBUTIONS
SVV, AV, LDJ, AW, ES, SD were involved in conception and design of the study. SVV drafted the
manuscript. SVV, AV, LDJ, AW, ES, SD revised the manuscripts critically for intellectual content and
read and approved the final manuscript. SVV, AV, LDJ, AW, ES, SD agree to be accountable for the
contents and integrity of this manuscript.

CONFLICTS OF INTEREST
None declared.

FUNDING
Ministry of Health grant number 318804; National organization for health research and
development.

138
ACKNOWLEDGEMENTS
We would like to thank the following institutions/ persons from the Erasmus Medical center: the
Departmen of Trials and Research Coordination, Dr. J.C. van der Woude (independent physician
for the (eligible) study population), Department of Public Health (F. Santegoets) for construction
and continuous support in the use of the Gemstracker system, Preconceptiewijzer (S. Horsman) for
facilitation with accounts and Clinical Chemists (Dr. R. de Jonge). We would also like to thank the
Erfocentrum for collaboration in the use of tools.
We would like to thank the following parties for participating in the sub-study: local project
managers in participating HP4All municipalities (M. Bunnik, M. van der Ham, M. van der Biezen,
G. Franssen, B. Zeelen, A. de Graaf, R. Stoffels, S. Wiggers, G. de Rijk, B. Heusschen, S. Badal, E.
Hochheimer, A. Verhoeff ), participating midwife practices and General Practices, preconception
health educators and participating Youth Health Care services. We thank all clinical chemists and
local trial coordinators of participating laboratories (Erasmus MC, ATAL-medial, Atrium MC, SHO,
Flevoziekenhuis, Certe, Labwest, MST, Saltro). We would like to thank the advisory board of the
HP4All study.

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28. Stichting Erfocentrum en Erasmus MC. Preconception questionnaire [ZwangerWijzer]. www.zwangerwijzer.nl.
29. Landkroon AP, de Weerd S, van Vliet-Lachotzki E, Steegers EA. Validation of an internet questionnaire for risk assessment in
preconception care. Public Health Genomics. 2010;13(2):89-94.
30. Vardavas CI, Chatzi L, Patelarou E, et al. Smoking and smoking cessation during early pregnancy and its effect on adverse
pregnancy outcomes and fetal growth. Eur J Pediatr. Jun 2010;169(6):741-748.
31. De-Regil LM, Fernandez-Gaxiola AC, Dowswell T, Pena-Rosas JP. Effects and safety of periconceptional folate supplementa-
tion for preventing birth defects. Cochrane Database Syst Rev. 2010(10):CD007950.
32. de Weerd S, Thomas CM, Cikot RJ, Steegers-Theunissen RP, de Boo TM, Steegers EA. Preconception counseling improves
folate status of women planning pregnancy. Obstet Gynecol. Jan 2002;99(1):45-50.
33. Castles A, Adams EK, Melvin CL, Kelsch C, Boulton ML. Effects of smoking during pregnancy. Five meta-analyses. Am J Prev
Med. Apr 1999;16(3):208-215.
34. Mullally A, Cleary BJ, Barry J, Fahey TP, Murphy DJ. Prevalence, predictors and perinatal outcomes of peri-conceptional
alcohol exposure--retrospective cohort study in an urban obstetric population in Ireland. BMC Pregnancy Childbirth.
2011;11:27.
35. van Gelder MM, Reefhuis J, Caton AR, et al. Maternal periconceptional illicit drug use and the risk of congenital malforma-
tions. Epidemiology. Jan 2009;20(1):60-66.

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36. Gemstracker. https://gemstracker.org/general-information/.
37. Connor Gorber S, Schofield-Hurwitz S, Hardt J, Levasseur G, Tremblay M. The accuracy of self-reported smoking: a sys-
tematic review of the relationship between self-reported and cotinine-assessed smoking status. Nicotine Tob Res. Jan
2009;11(1):12-24.
38. de Weerd S, Thomas CM, Kuster JE, Cikot RJ, Steegers EA. Variation of serum and urine cotinine in passive and active smok-
ers and applicability in preconceptional smoking cessation counseling. Environ Res. Oct 2002;90(2):119-124.
39. Greenfield TK, Kerr WC. Alcohol measurement methodology in epidemiology: recent advances and opportunities. Addic-
tion. Jul 2008;103(7):1082-1099.
40. Salaspuro M. Carbohydrate-deficient transferrin as compared to other markers of alcoholism: a systematic review. Alcohol.
Nov 1999;19(3):261-271.
41. Tavakoli HR, Hull M, Michael Okasinski L. Review of current clinical biomarkers for the detection of alcohol dependence.
Innov Clin Neurosci. Mar 2011;8(3):26-33.
42. de Walle HE, de Jong-van den Berg LT. Ten years after the Dutch public health campaign on folic acid: the continuing chal-
lenge. Eur J Clin Pharmacol. May 2008;64(5):539-543.
43. Bakker R, Kruithof C, Steegers EA, et al. Assessment of maternal smoking status during pregnancy and the associations with
neonatal outcomes. Nicotine Tob Res. Dec 2011;13(12):1250-1256.
44. Elsinga J, de Jong-Potjer LC, van der Pal-de Bruin KM, le Cessie S, Assendelft WJ, Buitendijk SE. The effect of preconception
counselling on lifestyle and other behaviour before and during pregnancy. Womens Health Issues. Nov-Dec 2008;18(6 Sup-
pl):S117-125.

6
45. CCKL. Coördinatie Commissie ter bevordering van de Kwaliteitsbeheersing op het gebied van Laboratoriumonderzoek in
de Gezondheidszorg CCKL. http://www.cckl.nl/index.php?pagina=72. Accessed May 5 2014.
46. The International Planned Parenthood Federation European Network (IPPFEN); Barometer of womens’ access to contracep-
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The HP4ALL preconception care sub-study

47. Cefalo R.C., Moos M.K. Preconceptional Health Care: A Practical Guide. St. Louis: Mosby-Year Book; 1995.

141
 PART III

3
IMPLEMENTATION
& EVALUATION
CURRENT PRACTICES OF
PRECONCEPTION CARE
BY PRIMARY CAREGIVERS
IN THE NETHERLANDS
S.F. van Voorst, S.C.N. Plasschaert, L.C. de Jong-Potjer,
E.A.P. Steegers, S. Denktaş
European Journal of Contraception and Reproductive
Health Care 2016; 21(3)
ABSTRACT
Objectives: Over the past decade the value of preconception care (PCC) consultations has been
acknowledged. Investments have been made to promote delivery and uptake of PCC consultations
in the Dutch primary care setting. We assessed current activities, perceptions and prerequisites for
delivery of PCC in primary care.
Methods: A questionnaire was compiled and distributed by mail or e-mail among 1682 general
practitioners (GPs) and 746 midwives in the Netherlands between 2013 and 2014.
Results: The questionnaire was completed by 449 GPs and 250 midwives. While GPs and midwives
were frequently asked about preconception risks, explicit requests by patients for a PCC consultation
were less frequent. Although caregivers gave information on preconception risk factors, only a
minority recommended PCC in the form of a dedicated consultation. Such consultations occurred
infrequently. Risk factor assessment varied between GPs and midwives. Respondents’ perceptions
of PCC consultations, however, were generally positive. A small proportion believed that PCC
medicalised pregnancy, and recognised barriers in actively raising the topic of patients’ pregnancy
wishes. More training, staff, promotion of PCC and adequate reimbursement were prerequisites
for future delivery. GPs differed in their opinion of whether they or midwives were primarily
responsible for PCC consultations. Midwives, however, saw themselves as responsible for providing
PCC consultations.
Conclusions: Primary care is underserving prospective parents with regards to PCC consultations.
Targets to increase delivery of systematic PCC are: (1) promotion during routine care; (2) increased
use of tools; (3) increased collaboration among primary caregivers; (4) reduction of caregivers’
negative perceptions; and (5) tailoring PCC consultations to suit women’s preferences.

146
INTRODUCTION
Preconception care (PCC) is care for all women or couples contemplating pregnancy that aims to
identify and modify biomedical, behavioural and (psycho)social risks to parental health and the
health of the future child through counselling, prevention and management.1 The number of PCC
risk factors is abundant.1,2 An example of a PCC measure applicable to every woman is folic acid
supplementation. PCC measures depend on the risk profile of the woman or couple. An example is
strict glycaemic control in the case of diabetes. Intervention before conception gives time to tailor
a PCC health plan to individual needs in order to optimally reduce risks before the critical phase
of placentation and organogenesis. This phase is crucial to the course of pregnancy and perinatal
health outcome. PCC has therefore been internationally recognised as a method to improve
perinatal health.
In the Netherlands, improvement of perinatal health is highly relevant. The perinatal mortality
rate in the Netherlands is high and has declined more slowly than in other European countries over
the past decade.3 PCC is regarded as a feasible measure with great potential to improve perinatal
health, because couples in the general Dutch population are known to have a high prevalence of
preconception risk factors but generally plan a pregnancy.4
PCC can be delivered in many ways: the ideal approach depends on the local health system.5
In the Netherlands, delivery of PCC in the form of an individual PCC consultation is advocated.6
Individual consultations provide the opportunity for professional-led broad risk assessment to 7
ensure that risk factors are not overlooked. Furthermore, it encourages the delivery of interventions
in a tailored fashion and monitoring of improvement in PCC health by a professional.

Current practices of individual preconception care

The effectiveness of PCC is debated. Evidence for PCC is mostly based on association studies of
preconception risks and the occurrence of adverse pregnancy outcomes. Theoretically, eliminating
risk factors should lead to improvement of preconception health (e.g., risk of maternal smoking
is avoided after smoking cessation). Although evidence has been established for many single
preconception interventions (e.g., folic acid supplementation), the effectiveness of an integrated
approach in which interventions are delivered as a set or programme has not yet been established.7
The introduction of individual PCC consultations has been advocated in the Netherlands since
2007, based on the available evidence for risk factors and evidence for single preconception
interventions.
As in other countries with strongly developed primary care settings, in the Netherlands general
practitioners (GPs) and midwives are seen as responsible for delivering individual PCC consultations
to the general public. Several prerequisites for delivery of PCC by GPs and midwives have been
met over the past decade in order to enable this. First, guidelines for individual PCC have been
developed.8 Second, different standardised risk assessment tools have been developed.9,10 Third,
different pilot projects in the GP and midwife settings have taken place which show positive
attitudes of Dutch women towards PCC consultations.11–13 Lastly, prior audits show positive
ambitions of GPs and midwives to deliver PCC. Despite the aforementioned developments, PCC
consultations remain scarce.14–16 No studies have, however, assessed what primary caregivers
actually do with regard to PCC consultations. This study therefore aimed to establish to what extent

147
Dutch GPs and midwives currently promoted and provided individual PCC consultations. The study
also aimed to evaluate caregivers’ perceptions about PCC and their prerequisites for future delivery.
These perceptions and prerequisites are potential targets to increase the delivery of individual,
standardised PCC consultations in primary care.

METHODS
Design and setting
A cross-sectional audit was conducted as a pre-intervention study prior to the implementation of
PCC consultations within the Healthy Pregnancy 4 All (HP4All) PCC substudy.17 The central aim of the
HP4All PCC substudy is to develop a standardised approach to PCC consultations. This standardised
approach requires GPs and midwives to perform PCC using a validated questionnaire and according
to protocols. The present study designed a survey to address current activities, perceptions and
prerequisites regarding delivery of PCC and was carried out among primary caregivers within the
50 municipalities identified at the launch of the HP4All PCC substudy. These municipalities were
identified because they have the highest perinatal mortality and morbidity rates in the country. The
municipality selection process is described elsewhere.18 The municipalities were categorised into
14 intervention municipalities and 36 non-intervention municipalities. The survey was carried out
as described below.
All midwife practices within the 50 municipalities were located through the midwives’
professional organisation. Practices were contacted and asked for individual contact information
of affiliated midwives. If provided, midwives were personally invited to participate; otherwise, the
contact person was asked to distribute the surveys among all midwives in the practice.
All GP practices were located within the 14 intervention municipalities and in a random sample
of 50% of the postcodes in the 36 non-intervention municipalities. This sample was drawn because
it was estimated that 50% of the postcodes would provide a sufficient number of respondents to
fulfil the aims of the study. Second, the sample was drawn for feasibility reasons: in the absence
of an up-to-date list of GP practices per postcode, locating practices would have involved a time-
consuming internet search. It would have been too onerous to perform an online search for all
postcodes. Similar to the procedure to recruit midwives, GP practices in the selected areas were
contacted and asked for individual contact information of affiliated GPs. If provided, GPs were
personally invited to participate; otherwise, the contact person was asked to distribute the
questionnaires among all affiliated GPs in the practice.

Data collection
The authoring team compiled a questionnaire of 23 open-ended or closed questions within three
domains: (1) respondents’ characteristics; (2) current practices assessed over the two months
prior to filling in the questionnaire; and (3) perceptions. The questionnaire was piloted to assess
whether it was understandable and covered all potential answer categories. This was done
by asking two GPs, a midwife and an obstetrician to fill in the questionnaire. Adjustments were

148
made accordingly. A summary of the questionnaire is presented in Figure 1; the full questionnaire
is available on request. The questionnaire was available on paper and via an internet link sent by
e-mail. Respondents were invited to participate by phone or by letter. In the case of non-response a
reminder was sent. Data collection was performed between February 2013 and February 2014. The
questionnaire was distributed prior to implementation of PCC in the intervention municipalities of
the HP4All PCC substudy.

FIGURE 1: Domains, constructs and items of the questionnaire.

Current practices of individual preconception care

149
Analysis
Results were analysed using SPSS 20.0 software (Statistical Package for Social Sciences, Chicago, IL,
USA) and descriptive statistics, and χ2 or Fischer’s exact test where applicable to test for significant
differences in proportions. Significance was defined as a p-value <0.05.

RESULTS
Respondents
Of the 1682 GPs, 449 filled in the questionnaire (individual response rate 27%). These responses
accounted for 268 of 763 GP practices (practice response rate 35%). Of 746 midwives, 250 filled
in the questionnaire (34%), accounting for 108 of 187 approached midwife practices (practice
response rate 58%). Table 1 presents the characteristics of the respondents. Respondents were
representative of Dutch GPs and midwives, except for a slight overrepresentation of female GPs,
part-time employed GPs, and self-employed midwives. PCC training was reported by 15% of GPs
and 63% of midwives; 20% of the GPs and 67% of the midwives rated their knowledge of the PCC
guideline as good (rather than moderate or not at all).

TABLE 1: Characteristics of the respondents.

Characteristic GPs (n=449) Midwives (n=250)

No. % Ref. No. % Ref.
Sex
 Male 205 45.7 57.4 4 1.6 1.6
 Female 236 52.6 42.6 244 97.6 98.4
Age in years, median (range) 47 (24-66) - 49 (27-87) 35 (21-65) - 36 (21-65)
Type of employment
 Self-employed 376 83.7 89.9 182 72.8 51.2
 
Employed by another self-em- 45 10.0 11.1 20 8.0 6.2
ployed GP/midwife
 Employed by a primary care prac- NA NA NA 24 9.6 2.3
tice or organisation
  Employed by a hospital NA NA NA 6 2.4 27.7
 Locum/temporary NA NA NA 14 5.6 12.7
Employment
  Part time 343 76.4 57.4 143 57.2 53
  Full time* 102 22.3 42.6 105 42.0 47.1
Type of practice
 Solo 114 25.4 25 8 3.2 5.4
 Duo 110 24.5 37.9 23 9.2 15.0
 Group 212 47.2 36.4 208 83.2 79.6
 Other 0 0 0 3 1.2 0

Percentages do not always add up to 100%, due to missing values.

Ref., reference characteristics of GPs and midwives in the Netherlands in 2012, provided by the Netherlands Institute for
Health Services Research (Nivel); NA., not applicable.*Full time is defined as 40 h per week.

150
Current PCC practices
Table 2 shows the current demand, offer and delivery of PCC consultations.

TABLE 2: Demand for and offer and delivery of PCC.

Variable GPs (n=449) Midwives (n=250) p-value*

No. % No. %
Demand for PCC
 Received a question about risk factors for po- 257 57.2 101 40.4 <0.005
tential pregnancy**
 Received an explicit request for a PCC consul- 104 23.2 69 27.6 0.183
tation**
Offer of PCC
 Pointed out a risk factor in a future pregnan- 299 66.6 107 42.8 <0.005
cy**
 Policy to bring a PCC consultation to patient’s 379 84.4 204 81.6 0.338
attention at an appropriate moment
  Explicitly recommended a PCC consultation** 74 16.5 54 21.6 0.086
 Systematically invited patients for a PCC con- 2 0.7 4 1.6 0.193
sultation (e.g. by direct mailing)

*χ2 test was applied; when data in cells were <5, Fischer’s exact test was applied.
**In the past 2 months.

7
Demand

Current practices of individual preconception care

Both GPs and midwives had been asked questions about preconception risks in the previous two
months: GPs more often than midwives (57% vs. 40%; p<0.005). There were fewer specific requests
for a PCC consultation (23% of GPs and 28% of midwives).

Offer
In the previous two months, 67% of GPs and 43% of midwives reported that they had mentioned
to patients risk factors for a future pregnancy. GPs did this significantly more often than midwives
(p<0.005). The majority of GPs (82%) and midwives (84%) routinely mentioned the availability of
PCC consultations during their clinical practice. Opportunities that both caregivers took to mention
the availability of PCC were if women mentioned a desire to become pregnant (66% of GPs and 66%
of midwives), during care after a miscarriage (45% of GPs and 54% of midwives), and when adverse
pregnancy outcomes were apparent (36% of GPs and 48% of midwives). Fifty percent of midwives
mentioned PCC consultations (or interconception care) during the routine postnatal check-up a few
weeks after delivery. Among activities in the daily practice of GPs, a majority reported the availability
of PCC during consultations about hereditary conditions (57%). Opportunities in general practice
that were reported to be taken by a minority of GPs were: prescription of a medication (25%), when
contraception was discussed (14%), and during routine follow-up of chronic medical conditions
(16%). A few GPs (52%) reported that they mentioned the availability of PCC if their patient was
getting married, feared encountering problems during pregnancy, requested travel vaccination,

151
was undergoing evaluation of chronic medication use or a Pap smear, and if sexually transmitted
infections or sexual matters were addressed. However, explicit invitation for a PCC consultation had
occurred less frequently in the two months prior to the questionnaire (by 17% of GPs and 22% of
midwives). Very few GPs and midwives systematically sent out invitations for PCC consultations to
women in their patient record system.

Delivery
A small proportion of GPs and midwives had carried out PCC consultations in the two months prior
the questionnaire (27% of GPs and 20% of midwives). The proportion of GPs who performed a PCC
consultation was significantly higher compared with the proportion of midwives.
Respondents were asked how they organised the delivery of PCC. Twenty percent of GPs (n = 91)
and 49% (n = 123) of midwives reported providing PCC consultations themselves according to their
professional guideline (i.e., their PCC constituted a risk assessment across the domains presented in
Figure 2). We restricted our analysis to the content of PCC reported by these respondents. Figure 2
presents the PCC risks that are routinely assessed by respondents who reported carrying out PCC
consultations themselves. Pap smears, eating disorders, vitamin A, low body mass index, rubella
immunisation, work exposures and stressors were assessed by <40% of GPs. Domestic exposures,
presence of uterine anomalies, and risks due to travel received less attention from both GPs and
midwives. For the majority of risk factors, a significantly larger proportion of midwives reported
assessing them compared with GPs. This could be inherent to the fact that GPs are the medical file
keepers within the system. Content of delivered PCC is also influenced by the use of tools such as
screening question- naires, as recommended in guidelines. Of those included in the analysis shown
in Figure 2, 25% of GPs and 94% of midwives reported using a tool for delivery of PCC consultations.
The tools they reported using were the web-based questionnaire ZwangerWijzer19 (12% of GPs
and 83% of midwives), its complementary archive software programme, PreconceptieWijzer9
(12% of GPs and 11% of midwives), the questionnaire provided by the professional organisation
of midwives (39% of midwives), a self-assembled intake form (1% of GPs and 3% of midwives), or a
questionnaire integrated into the patient record system (2% of midwives).

152
FIGURE 2: E
 lements of PCC and proportion (%) of GPs (n=91) and midwives (n=123) who included these
risk factors in their standardised PCC consultation.

Obstetic / gynaecologic history

obstetric history *
prior gynaecological
surgery *
pap smaer *
uterine anomalies *

Maternal diseases
maternal diseases

Nutrition, dietary supplements

eating disorders *
high body mass index
low body mass index *
diets [1] *
folic acid supplements
vitamin A *

Medication
prescribed medication
over the counter drugs

Immunization
7
rubella immunization *

Current practices of individual preconception care

Infectious diseases
exposure to infectious
agents [2]
sexually transmitted
diseases *
Genetic diseases
ethnicity *
consanguinity *
family diseases:
hereditary diseases *
family history:
congenital abnormalities *
Alcohol, smoking, illicit substances
alcohol
smoking
drugs
domestic toxic exposures

Environmental exposures
work related toxic
exposures or radiation *
workrelated stressors *

Special groups
age > 36
travelling

0% 50% 100%
General Practitioners Midwives

153
Current perceptions about PCC
Figure 3 presents the agreement of respondents with statements about PCC. It shows that the
majority of respondents had a positive attitude towards PCC. Potential views that could be a barrier
to the delivery of PCC by GPs were that PCC consultations should only be offered to women with
high risks (30%), that PCC medicalised the preconception period (31%) and that offering PCC
without women asking for it was objectionable (23%). Twenty-three percent of midwives also
agreed with the last statement.

FIGURE 3: Views regarding PCC among GPs and midwives (MW).

PCC can lead to better pregnancy outcomes

GP

MW

The risk profile of my patient will not change after PCC

GP

MW

I prefer to deal with risk factors before pregnancy, rather than in pregnancy
GP

MW

Other information sources make PCC consultations obsolete

GP

MW

PCC should only be offered to women with high risks

GP

MW

Initiating the talk about pregnancy wish is uncomfortable

GP

MW

PCC leads to medicalization of the preconception period

GP

MW

PCC advice without women asking for it is objectionable

GP

MW

With PCC I can do something extra for patients

GP

MW

0% 50% 100%

GP = General Practitioner MW = Midwives Agree Neutral Disagree No response

154
Perceptions about PCC in the future
Respondents said they were willing to mention the availability of PCC during routine care, if they did
not already do so. They did not, however, favour discussing PCC during contraception counselling.
The majority of caregivers who did not use a tool would be willing to use one in the future (90% of
GPs and 71% of midwives).
Figure 4 shows the prerequisites for PCC delivery in the future. All items were prerequisites
for a substantial proportion of GPs and midwives. Respondents especially agreed that adequate
reimbursement and more promotion of PCC were prerequisites. Respondents were asked which
caregivers (GPs, midwives, gynaecologists or adolescent health care physicians) should be primarily
responsible for systematic delivery of PCC consultations. Among midwives, the majority (67%)
thought that midwives were primarily responsible. There was disagreement among GPs, as 42%
thought that midwives should be primarily responsible for delivery of PCC and 40% thought that
GPs should be responsible for its delivery. The remaining GPs and midwives thought that adolescent
health care professionals and gynaecologists were primarily responsible for the delivery of PCC.

FIGURE 4: Prerequisites for delivering PCC in the future among GPs and midwives (MW).

7
Evidence for effectiveness
GP

Current practices of individual preconception care

MW

Background knowledge
GP

MW

More staff
GP

MW

More promotion of PCC

GP

MW

Adequate reimbursement
GP

MW

0% 50% 100%

GP = General Practitioner MW = Midwives Agree Neutral Disagree No response

155
DISCUSSION
This audit shows that activities of GPs and midwives in PCC delivery mostly revolve around
answering questions and pointing out risk factors when asked by a patient. The step to a dedicated,
standardised PCC consultation is made less frequently. Approximately one in four GPs, and one
in five midwives, had given a PCC consultation in the two months prior to the survey. Given the
total number of pregnancies in the Dutch perinatal registry within the selected regions in 2013
(72,591 births in the postcodes of invited midwife respondents, 35,186 births in the postcodes
of invited GP respondents), the potential population for PCC in the two months could have been
13 women per midwife or 3.5 women per GP (assuming a planned pregnancy rate of 80% and an
equal distribution of conceptions throughout the year). In practice, however, the potential number
of PCC consultations in the GP setting is likely to be higher, as GPs have more contact with non-
pregnant women and opportunities in daily practice to address PCC compared with midwives.
Half the midwives and approximately 20% of GPs performed PCC in a standardised manner. We
conclude that only a minority of couples contemplating pregnancy are currently being offered PCC
consultations.
We believe that the strength of this study lies in the assessment of performed activities during
a set time period. These activities may be viewed in light of the demand caregivers receive and
how they promote PCC. A difficulty in assessing PCC activities is that caregivers have different
understandings of the content of PCC. Therefore, we first chose to assess the extent to which
PCC activities were performed according to caregivers’ own definition of PCC. We then chose to
assess the proportion of caregivers who conducted PCC systematically as stated in the guidelines.
Applying this definition in an earlier phase would have underestimated PCC activities. On the other
hand, we regret that we could not assess the actual performance of systematic PCC consultations
and the content of PCC delivered by caregivers that did not adhere to the guidelines. Other
recurring reasons for non-response were personal factors, a policy not to participate in studies in
general or from a non-affiliated centre.
We cannot exclude the presence of a selection bias, as it is feasible that caregivers with a
higher affinity for PCC were more motivated to participate in the survey. Caregivers’ interest
might have been influenced by simultaneous conversations about participation in the HP4All PCC
substudy that took place in 14 of the municipalities.17 These municipalities provided 36% of the GP
respondents and 53% of the midwife respondents, respectively. Response rates were 35% among
GPs and 56% among midwives in the HP4All municipalities vs. 23% of GPs and 21% of midwives in
the remaining municipalities. Subgroup analysis was performed to ensure that the design did not
affect the results. There were no significant differences in actual activities regarding PCC.
A limitation in our design was that we relied on self-reported delivery of PCC consultations.
Research in medical files would have been more reliable but was not feasible.
Previous studies in the Netherlands regarding delivery of PCC have been conducted before
the advocacy of individual, standardised PCC by the Dutch health board in 2007.6 The aim of these
studies was mainly to assess perceptions and attitudes about delivery of PCC among GPs and
midwives. The results showed that GPs and midwives occasionally provided a recommendation

156
about a single PCC risk.14–16 The studies, however, do not provide data about the frequency of PCC
activities and the extent to which PCC consultations were systematic. Therefore, we cannot reflect
on whether delivery of standardised PCC consultations has changed over time.
Comparison of activities of primary caregivers in other countries is limited to a few studies.20–23
Again, PCC in these studies seems to be limited to provision of one or more single pieces of advice
rather than a standardised, dedicated and systematic consultation. PCC should be seen in light
of countries’ policies. The Netherlands might be unique in its clear advocacy of standardised PCC
consultations in primary care by the Dutch health board and in professional guidelines for GPs
and midwives.6,8,24 This possibility is supported by a recent review of PCC policy in six European
countries.25
Other studies report the number of pregnancies exposed to PCC.26–28 In our opinion, this
number does not reflect implementation of PCC by caregivers because this number only reflects a
part of the actual delivery of PCC. It does not include PCC received by couples who did not conceive
or whom were offered PCC but did not utilize the service. In order to assess overall PCC activities we
advocate evaluation from the point of view of both delivery and receipt.
We recommend increasing PCC activities. With regard to everyday practice, GPs and midwives
should be more proactive and explicit about the availability of PCC consultations during appropriate
moments in routine care. As midwives have fewer opportunities in daily practice to inform non-
pregnant women about PCC than GPs, we recommend that GPs and midwives collaborate. This 7
could also be a solution for GPs who do not deliver PCC themselves. Increasing the use of tools can
promote uniformity of PCC consultations.

Current practices of individual preconception care

Training, reimbursement, more staff resources and recruitment strategies are prerequisites that
should be met. Among prerequisites, more evidence for the effectiveness of PCC was mentioned.
This perception is in contrast with the abundant amount of evidence for preconception risk factors,
which prompted the Dutch health board to decide that individual PCC should be delivered. Another
perspective could be that it is unethical not to inform prospective parents about preventive
measures. Training, guidelines and advocacy to deliver PCC by a professional organisation may
reduce negative perceptions about the effectiveness of PCC.

Future research
The difficulty of making changes in everyday practice should not be underestimated. We
recommend monitoring the implementation of standardised PCC as it finds its way to common
practice. This implementation research should aim to identify facilitators for and barriers to the
delivery of standardised PCC in the context of the health care system. Additionally, research is
necessary to align caregivers’ approaches to standardised PCC to the preferences and needs of
women. This could promote its uptake and therefore reward caregivers’ efforts, providing a positive
feedback loop.
This study was confined to PCC in the form of individual PCC consultations. Individual PCC
consultations have the advantage that thorough risk assessment across all risk domains can be
performed for a couple contemplating pregnancy. Yet, there is a trend internationally to integrate

157
PCC into well-women’s health care services. Although this is outside the scope of the current
study, we recommend that future research addresses how PCC can be integrated into preventive
health care services for women. This will require increased collaboration between the health care
prevention and primary care sectors.

CONCLUSIONS
Delivery of PCC to couples in the general population has been advocated since 2007. This study,
however, confirms that delivery of PCC only occurs for a minority of women contemplating
pregnancy. Targets to extend delivery of PCC are: (1) explicit promotion of comprehensive PCC
consultations at appropriate moments in everyday clinical practice; (2) promotion of standardised
content of PCC by increasing the use of tools; (3) collaboration between GPs and midwives to
promote and deliver PCC; (4) changing negative perceptions about PCC among GPs and midwives;
(5) improving uptake by tailoring PCC consultations to meet the needs of women.

CONFLICTS OF INTEREST
No funding was received for this study. The authors have no conflicts of interest to declare.

ACKNOWLEDGEMENTS
We thank J.M.W.M. Merkus and R. Hop for reviewing the questionnaire, and R.J. Kenens of the
Netherlands Institute for Health Services Research (Nivel) for providing reference statistics.

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tions, evidence and recommendations. J Community Genet. 2015;6:107–115. 

2. Jack BW, Atrash H, Coonrod DV, et al. The clinical content of preconception care: an overview and preparation of this sup-
plement. Am J Obstetr Gynecol. 2008;199(6 Suppl. 2):S266–279. 

3. EURO-PERISTAT. PERISTAT II study. European perinatal health report. Better statistics for better health for pregnant women
and their babies in 2004. 2008. [Internet]. [cited 2016 Feb 7] 2008. Available from: https://www.europeristat.com/images/
docEPHR/ european-perinatal-health-report.pdf.
4. van der Pal-de Bruin KM, le Cessie S, Elsinga J, et al. Preconception counselling in primary care: prevalence of risk factors
among couples contemplating pregnancy. Paediatr Perinat Epidemiol. 2008;22:280–287.
5. Boulet SL, Parker C, Atrash H. Preconception care in international settings. Matern Child Health J. 2006;10(5 Suppl.):S29–35.
6. Health Council of the Netherlands: Preconception care: a good beginning. Health Council of the Netherlands. The Hague
2007: publication no. 2007/19E, ISBN 978-90-5549-657-0. 

7. Temel S, van Voorst SF, Jack BW, et al. Evidence based preconceptional lifestyle interventions. Epidemiologic Rev.
2014;36:19–30. 

8. de Jong-Potjer L, Beentjes M, Bogchelman M, et al. Preconception care. NHG guidelines, 2011. [Internet]. [cited 2014 May
15].
9. Erfocentrum, Erasmus MC. PreconceptieWijzer [Preconception consultation tool for preconception care providers]. [Inter-

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10. Landkroon AP, de Weerd S, van Vliet-Lachotzki E, Steegers EA. Validation of an internet questionnaire for risk assessment in
preconception care. Public Health Genomics. 2010;13:89–94. 

11. Temel S, Birnie E, Sonneveld HM, et al. Determinants of the intention of preconception care use: lessons from a multi-ethnic
urban population in the Netherlands. Intl J Public Health. 2013;58:295–304.
12. Elsinga J, de Jong-Potjer LC, van der Pal-de Bruin KM, et al. The effect of preconception counselling on lifestyle and other
behaviour before and during pregnancy. Womens Health Issues. 2008;18(6 Suppl.):S117–125. 

13. Denktas S, Bonsel GJ, Steegers EA. [Perinatal health in Rotterdam, the Netherlands – experiences after 2 years of ’Ready for
a baby’]. Ned Tijdschr Geneeskd. 2012;156 (29):A4289.
14. Gaytant MA, Cikot RJ, Braspenning JC, et al. [Preconception counselling in family practice; a survey of 100 family physi-
cians]. Ned Tijdschr Geneeskd. 1998;142:1206–1210.
15. Poppelaars FA, Cornel MC, Ten Kate LP. Current practice and future interest of GPs and prospective parents in preconcep-
tion care in The Netherlands. Fam Pract. 2004;21:307–309.
16. van Heesch PN, de Weerd S, Kotey S, Steegers EA. Dutch community midwives’ views on preconception care. Midwifery.
2006;22:120–124.
17. van Voorst SF, Vos AA, de Jong-Potjer LC, et al. Effectiveness of general preconception care accompanied by a recruitment
approach: protocol of a community based cohort study (the Healthy Pregnancy 4 All study). BMJ Open. 2015;5(3):e006284.
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birth. 2014;14:253.
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www.zwangerwijzer.nl
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23. Best Start Resource Centre. Preconception health: physician practices in Ontario. Toronto, ON: Best Start 2009.

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Reprod Health Care. 2015;20:77–87.
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2004– 2010. Birth. 2014;41:367–373.
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159
DEVELOPING SOCIAL MARKETED
INDIVIDUAL PRECONCEPTION
CARE CONSULTATIONS:
WHICH CONSUMER PREFERENCES
SHOULD IT MEET?
S.F. van Voorst, C.A. ten Kate, L.C. de Jong – Potjer,
E.A.P. Steegers, S. Denktaş
Health Expectations 2017;1–8
ABSTRACT
Aims: Preconception Care (PCC) is care that aims to improve the health of offspring by addressing
risk factors in the pre-pregnancy period. Consultations are recognized as a method to promote
perinatal health. However, prospective parents underutilize PCC services. Uptake can improve if
delivery approaches satisfy consumer preferences. Aim of this study is to identify preferences of
women (consumers) as a first step to social marketed individual PCC consultations.
Methods: In depth, semi-structured interviews were performed to identify women’s views regarding
the 4 components of the social marketing model: product (individual PCC consultation), place
(setting), promotion (how women are made aware of the product) and price (costs). Participants
were recruited from General Practices and a midwife’s practice. Content analysis was performed by
systematic coding with NVIVO software.
Results: The 39 participants reflected a multiethnic intermediately educated population. Product:
Many participants had little knowledge of the need and the benefits of the product. Regarding
the content of PCC, they wish to address fertility concerns and social aspects of parenthood.
PCC was seen as an informing and coaching service with a predominant role for health care
professionals. Place: The General Practitioner and midwife setting was the most mentioned setting.
Promotion: A professional led promotion approach was preferred. Price: Introduction of a fee for
PCC consultations will make people reconsider their need for a consultation and could exclude
vulnerable patients from utilization.
Conclusion: This study provides consumer orientated data to design a social marketed delivery
approach for individual PCC consultations.

162
INTRODUCTION
Preconception care (PCC) includes all measures taken before conception to increase the health of
the prospective mother (parents) and child. It addresses risks associated with adverse pregnancy
outcomes. The large number of acknowledged preconceptional risk factors can be categorized
into 13 domains: health promotion, immunizations, infectious diseases, chronic medical
conditions, psychiatric conditions, maternal exposures, genetic risks, medication, nutrition,
environmental risks, psychosocial stressors, reproductive history and special groups.1,2 Whilst some
risks and interventions are applicable to all couples (e.g. lifestyle recommendations, folic acid
supplementation), some risks are only present amongst some individuals (e.g. a positive family
history for hereditary diseases).
PCC has been acknowledged as a valuable addition to perinatal health care, to improve and
reduce inequalities in perinatal health and women’s health.3,4 Many countries are facing challenges
regarding which approach for the delivery of PCC is best suited to their health care setting. In
the Netherlands, the Dutch Health Council advocates PCC for the general public in the form of
individual consultations.3 Rationale is that the majority of couples in the general population is
known to have at least one risk factor for which PCC would be useful.4 Furthermore a consultation
with a health care professional provides the opportunity for individual risk assessment and
intervention. However, despite availability of tools and guidelines, PCC consultations are only
offered at a small scale.5 When offered, uptake is low due to hesitancy amongst people to utilize
PCC.6,7 In order to increase the utilization of individual PCC consultations we need to address the
question of how this service should be delivered in order to meet demands and preferences of 8
prospective parents. Using a consumer-oriented approach to change behavior of a target group
(namely uptake of PCC services) is the basis of social marketing. Social marketing is defined as

towards social marketed preconception care

“the application of commercial marketing technologies to the analysis, planning, execution, and
evaluation of programs designed to influence voluntary adoption of recommended behaviors by
a targeted audience in order to improve their personal welfare and that of society.”8 One of the
steps is applying a marketing mix in which ‘product’, ‘price’, ‘place’, and ‘promotion’ characteristics
are blended in a marketing plan that reflects the appropriate mix of these 4 ‘P’s. The right ‘product’
has to be backed by the right ‘promotion’ and put in the right ‘place’ at the right ‘price’.9
Social marketing has been suggested to develop approaches for the delivery of preconception
care.10,11 As the Dutch health system advocates delivery of PCC in the form of individual PCC
consultations, this study is confined to the ‘product’ of individual PCC consultations. Goal of
the product is primarily to promote a healthy pregnancy and to reduce the chances of adverse
pregnancy outcomes. A consultation constitutes a thorough risk assessment to identify risks that
warrant intervention or counselling in the preconception phase. ‘Promotion’ concentrates on
the promotion of individual preconception care. The 3rd P, ‘place’ addresses characteristics of the
setting. The 4th P, ‘pricing’, includes the costs for patients for this product.
Aim of this study is to identify consumers’ preferences regarding these marketing components
as a first step in designing a socially marketed delivery approach for individual preconception care.

163
METHODS
This study is a prospective, community based, qualitative study.
Participants were enrolled via purposive sampling from waiting rooms at 2 general practices
(GP) and 1 midwife practice participating in the Healthy Pregnancy 4 All study.12 Staff of the practice
asked women if they would allow for a medical student to explain a study in which they could
participate. These women attended their respective practices for a scheduled appointment for
other health issues. If women were open to talk about participation in a study, a medical student
(CtK) explained the study and assessed the participants’ eligibility. Women in the reproductive age
range (18-42 years) who did not exclude a future pregnancy were eligible (see supplementary file
for script). Insufficient proficiency of the Dutch or English language was defined as an exclusion
criterion. If women agreed to participate they filled in a questionnaire on baseline characteristics
and the interview was scheduled at a convenient time at the respective practice. Sample size was
set at 40 interviews. Fewer interviews were deemed sufficient if theoretical saturation would be
reached at an earlier point.
Data collection consisted of individual semi-structured interviews. The topic list was designed
to address each ‘P’ of the marketing mix. Questions were formulated to identify aspects of the
4’ps which authors had brainstormed to be important and which are known to be of importance
in literature. As the interviews proceeded the interview strategy was adapted slightly, to ensure
that participants understood the questions. The topic list contained 27 open-ended questions
- with scripted sub questions when relevant – (Supplementary file 1). In order to ensure
successful discussion about individual PCC, we provided a definition of our product: individual
PCC consultations. All interviews were recorded and transcribed verbatim. Participants filled in a
questionnaire on baseline characteristics. The ethics committee approved the study (MEC 2013-
586). All participants provided informed consent for the recording and the use of data.
The interview transcripts were analyzed to identify elements of the social marketing model.
Analyses were done with NVIVO software for qualitative analysis of data.13 After the data was
imported, a basic coding scheme was made according to each P of the social marketing model.
This coding scheme was piloted. Two researchers independently applied the coding scheme to 10
interviews, and discussed discrepancies and modification of the nodes to optimally fit the content
of the interviews. This led to a definitive codebook. The remainder of the interviews were coded by
one researcher and checked by the other researcher. With the matrix coding function and query
function of NVIVO, contents could be analyzed to identify contents (perceptions of respondents)
and patterns in contents (consistency, frequency). Quotes were extracted to illustrate findings. The
quotes were translated from Dutch to English (by a native speaker) and back again (by a second
translator) to verify consistency of the translation.

164
RESULTS
40 women were recruited. One interview was discontinued because the candidate did not speak
Dutch or English sufficiently to understand and answer the questions. After the 36th interview, no
new information was provided and it was decided to stop data collection after 39 interviews. 23
participants were recruited from the midwifery setting, 16 participants were recruited from the
GP setting. Mean interview time was 22 minutes. Table 1 presents the characteristics of the study
participants. Participants were between 21 and 38 years old and reflect a multiethnic, intermediately
educated population. At the time of the interview 56% of the participants were pregnant. Most
non-pregnant participants did not contemplate pregnancy within the next 6 months.

Product

TABLE 1: Characteristics of participants.

Baseline characteristics of participants N (%) Total = 39

Age Median age (years) 27.97 (21-38)
Obstetric history Nulliparous 19 (48.7)
Multiparous 20 (51.3)
Maternity 0 children 25 (64.1)
1 child 10 (25.6)
2 children 3 (7.7)
3 children 1 (2.6) 8
Current pregnancy wish pregnant at the moment 22 (56.4)
planning pregnancy <3 months 1 (2.6)

towards social marketed preconception care

planning pregnancy 3-6 months 0
planning pregnancy >6 months 16 (41.0)
Marital status Married 22 (56.4)
Cohabiting 9 (23.1)
In a non-cohabiting relationship 5 (12.8)
Single 3 (7.7)
Ethnicity1 Dutch 26 (66.7)
Surinamese 2 (5.1)
Turkish 1 (2.6)
Moroccan 3 (7.7)
Other 7 (17.9)
Educational attainment level2 Low 4 (10.3)
Intermediate 17 (43.7)
High 16 (41.0)
Other 2 (5.0)

Numbers reflect number of participants (N) unless specified differently. (1) Ethnicity is defined as the social or cultural
group that the participant considered themselves to be part of; (2) Educational attainment level was classified according
to the International Standard Classification of Education (ISCED).33

165
 Knowledge about the purpose and the contents of PCC consultations was low amongst
participants. The majority presumed PCC to be fertility related care. Its aim was ‘to help women get
pregnant’, hastened by many participants with ‘as fast as possible’ or ‘within the desired time frame’.
Participants also thought its goal would be to help women with decisions about parenthood. In line
with these presumed goals, participants mentioned that the target group would consist of women
in the prepregnancy period ranging from women considering having a child to subfertile women.
In line with this, presumed content would be education about fertility and diagnostic work-up and
treatment of subfertility.
After participants were informed about what PCC actually was, intentions to utilize PCC varied.
Reported reasons to utilize PCC were mostly to be informed on their questions about perceived
risks and fertility. Participants reported they would be more likely to utilize PCC consultations after
trying to become pregnant for a longer period or when they are becoming pregnant for the first
time. Multiparity was reported as a reason not to utilize PCC, because most participants thought
they would know enough after prior pregnancy experiences. Lack of perceived need or benefit of a
PCC consultation was the most recurrent theme, as one respondent said illustratively:
“I still believe my body protects the fetus against harmful exposures during the first
three months. Secondly, it has been going fine without the existence of PCC services
in the past, so it will be fine regardless.”
Practical considerations (e.g. having to take time off from work), having other information sources
or feelings about interference in the privacy and spontaneity of conception were other reasons not
to utilize PCC consultations.
Regarding the contents of PCC, participants preferred PCC to address fertility, questions about
their perceived risks and about parenthood. In line with this the most mentioned approach for the
consultation was the provision of information. A few participants mentioned a preference for a
coaching approach:
“You can stop with contraception; however it would be better if you were coached

in the course of becoming pregnant instead of – ‘well ok I’m just going to stop with
contraceptives, and just see what happens.”
Although contact with peers and the use of tools (apps, internet, questionnaires) were valued
positively, participants valued them as an addition to professional lead PCC consultations rather
than a replacement. The personal approach, the authority and the credibility of a health care
professional were the most important advantages of a PCC consultation by a professional.
Participants mentioned the lack of credibility of the information and privacy issues, as the main
disadvantage of forementioned tools. A hallmark for tools with trustworthy information sources
and a function in tools where questions could be placed for answering by a health care professional
were suggested improvements.
Expertise, trust and involvement in care for pregnant women were mentioned as the
most important prerequisites of PCC providers. Based on these attributes GP’s, midwives and
gynecologists were most frequently suggested as PCC providers. Delegation of care to a nurse/
nurse practitioner/ medical assistant within general practices was deemed appropriate.

166
 CONSUMER PREFERENCES TO INDIVIDUAL PRECONCEPTION CARE CONSULTATIONS

Product Promotion Place Price

Product · Low knowledge about goals, contents, Media 1. By health professionals: Where Preferences: Willing- Preferences:
Knowledge benefits of PCC · by GP’s or midwifes · Accessibility by transport, closeby, private ness to · Substantial proportion would not be willing
· Low risk perception · Text message/ emails/ letters from place, place close to other services that pay to pay
GP’s might be necessary in the PCC phase or Willingness to pay depends on:
2. Anonymous aproaches for antenatal checkups, relation with the · Perceived need
Intention toFactors promoting uptake: · Leaflets subject pregnancy/ health, familiar place, · Satisfaction with alternative information
utilize PCC · Perceived need for information or lack of · Posters not to formal, place where other women services
satisfaction after other information sources · TV come · Perception that health insurance costs
· Perceived personal risks · Newspaper adds Proposed places: alot of money: it shoudl be included
· Fertility problems 3. Communication within the target group · midwife practice, GP practice, community · Personal financial situation
· Mouth to mouth location, hospital, healthcenter, · Attitude to everything within ones reach
Factors impeding uptake: 4. Information sources gynaecologist department, municipal for a healthy pregnancy
· Time restraints to visit a provider · Websites health service, own home, library
· Preference for other information sources · Books
(internet, peers)
· Prior pregnancy (provides information and
experience)
· Perceived low benefit/ necessity When In daily life: Prices Maximum price:
· Not planning pregnancy · Health care settings · Median 20 [minimum 0, maximum 88)
· Confrontation with risks · Places with a relation to the subject When Preferences:
· Privacy issues (e.g. maternity stores) or existing · flexibility to workschedule, willingness to
· Embarrassment peer groups (e.g community centers, take time off from work for daytime
· Unfamiliarity with PCC day care centers) consultations varies

When to target women: Proposed moments:

· When they start contemplating · during the day, evening, in the weekend
Content/ · Receiving information about ferility and
pregnancy or when they are trying to

167
approach do’s and don’ts
conceive.
· Adressing perceived personal risks
· Fertility problems
· Peers are not a replacement for
professional lead risk assessment · Prior adverse birth outcomes
· Place of digital tools is in addition to
availability of PCC consultations Profess- Who:
ionals · GP’s
Who Preferences: · Midwives
· Characteristics: expertise, personal known
caregiver, role in care for pregnant Target group:
women, access to medical record, · General approach; regardless of
potential to refer, easy to make an risks
appointment, good in advising or
explaining things Moments in clinical practice
· Main candidates: GP’s (or practice nurse/ · Pregnancy wish, fertility matters,
assistent), midwives, gynaecologist negative pregnancy test, initiation of
· Other mentioned candidates: maternity contraception, hereditary diseases,
nurse, multidisciplinairy team, youth health prescription of new medication, care
care nurse, fertility doctor, social worker after miscarriage, final consultation 6
weeks postpartum
· (See figure 2 for perceptions about
these moments)

Translation to socially marketted approach for individual PCC

consulations
FIGURE 1: P erceptions and preferences of women regarding the four components of social marketing model:
Product, Promotion, Place and Price. Items are listed according to the frequency they were mentioned.

towards social marketed preconception care

8
Promotion:
Four communication approaches to make women aware of PCC were mentioned by participants
(see Figure 1). The most preferred way to be informed about PCC was through a professional, mostly
directly or indirectly via an email, text message or a letter. GP’s were seen as the most suitable
professionals to do so as they are the starting point for health care in the Dutch Health system and
everybody has a GP. Midwives were also seen as suitable professionals to promote PCC. However,
participants mentioned that people generally associate midwives to care during pregnancy. Figure
2 displays perceptions about the suitability of contact moments with GP’s and midwives to be
informed about PCC.
In the promotion of PCC, participants preferred a general approach in which professionals
promote PCC to all women so everybody would be enabled to make an informed decision whether
or not they would utilize PCC. Suitable places for the promotion of PCC were all related to either
pregnancy or the target group. Participants preferred to be made aware of PCC when they start
thinking about becoming pregnant or when they are trying to become pregnant. They mentioned
that this is most likely when they have a stable life, being married or having finished education.
There is understanding that these factors differ per person and that early promotion of PCC is
necessary to reach women in time. Participants realized that caregivers generally do not know
whether women are planning a pregnancy or not.

Place
Accessibility, in terms of distance and convenience with public transport, was the most important
prerequisite. Other recurrent preferences of the location were privacy, location close to other
services related to PCC (e.g. access to midwifery care or dietician if needed), familiar places or
places where other women would come. These attributes caused participants to mention primary
care places (midwifery practices, GP practices, health centers) or hospitals (where gynecologist/
specialist care takes place) as suitable settings for a PCC consultation. At home, municipal health
centers and community centers were also mentioned.
Flexibility to consumers’ working schedules was the most mentioned prerequisite regarding
time. With differences in willingness to take time-off from work between participants, consultations
in the evening or even in the weekend were mentioned to be preferred or even essential to some.

168
FIGURE 2: S
 uitability of moments for health care providers to promote a preconception care consultation –
according to participants. Participants were asked to rate (grades 1- 10) the suitability of moments
in routine care for a health provider to point out the opportunity to have a PCC consultation. Based
on these grades, moments were ranked from being most suitable (top, green) to being least suitable
(bottom, red).
Pregnancy wish + Ideal moment to be informed
It is likely that there is a pregnancy wish, which
Fertility matters + is the ideal moment to be informed about PCC
consultations
It is likely that there is a pregnancy wish, which
Negative pregnancy
test
+ is the ideal moment to be informed about PCC
consultations
Contraception has to do with pregnancy/
+ contraception has a direct relationship with a
Initation of contracep-
pregnancy wish in the future
tion
– Might not be open for PCC
+ Relevant topic for future offspring
Hereditary diseases
– Confronting and scary
Acceptable if the specific drug influences fu-
± ture pregnancy or the health of the child, yet
risk perception regarding drug varies
± Less suitable if there is no pregnancy wish
Prescription of new
medication
Mentioning safety of the drug before/ during
– pregnancy on the box or the insert of the drug
is sufficient
Drugs are needed to improve the women’s
– health first, which is less important than the
pregnancy at that moment
Information about becoming pregnant or pre-
+ venting a miscarriage is likely to be welcome
Care after a miscarriage
Time is needed before parents have an open
– mind about the next pregnancy
You can be informed if you did not know
+ about PCC before the respective pregnancy.
If you mention PCC it can be related to risks
+ that became apparent in the respective preg-
6 weeks post-partum nancy/ delivery.
Suitable in case of problems during pregnan-
cy and labour; might be less suitable because
± women are not thinking about the next preg-
nancy at that moment
169
“At that moment you are disclosing that you have a
pregnancy wish, that is the moment when you need
the information”
“I wouldn’t be bringing it up if I would not be interest-
ed in having a child”
“It depends on if that person has a pregnancy wish.
But if that person does want to become pregnant,
then they can get advice.”
“Suitable because there is a relation between having
children and contraception”; “When you stop some
types of contraception, you can get pregnant imme-
diately. With other types it takes a while before you
become pregnant. It’s good to know this on forehand,
for the future.”; “If you stop, chances are big you want
to become pregnant.”
“When you discuss contraception you want to talk
about not becoming pregnant.”
“It is relevant for pregnancy and for your future child”
“No, that doesn’t feel good. No I would get extra wor-
ried then.”
8
“If the medication has low risks, for example in case of
Astma, then it’s not necessary. I would find it weird if
my GP would mention it. However I would want to be
informed if medication would have more risks.”
towards social marketed preconception care
“Ok if I would be wanting to become pregnant at that
moment, however if I wouldn’t I wouldn’t find it ac-
ceptable.”
“It is not necessary because it says so on the box or the
insert of the prescription.”
“If you are sick, you need something to get better first”
“Because I have had a miscarriage myself. At that time I
thought: how will I become pregnant again and which
advices should I be following? So that would be a ra-
tional moment.”
“It took me a long time before I was open to talking
about the miscarriage”
“Because if you didn’t have a PCC consultation before
the respective pregnancy, you can be told about it so
you now about it in case you want to become preg-
nancy again.”; “You may have questions related to you
prior pregnancy”
“If it would be relevant. But if everything went well
during the pregnancy and labour, I don’t see the need
of pointing out PCC”; “I don’t think that would be what
is on you mind then”
Price
Willingness to pay is mostly related to own financial situations and perceptions about
reimbursement of health care in the Dutch system - where health insurance is mandatory and
perceived as expensive. The requirement to pay for PCC would make a substantial proportion of
participants seek other (free) alternatives for a PCC consultation or to reevaluate their need for a
PCC consultation. This could provide a dilemma, for instance to women on social benefits.
“Just financially speaking, if it is not reimbursed, it would not be convenient, because
I am on social welfare, I have fixed expenses, and sometimes at the end of the month
it’s difficult to pay them and I have to stick it out. My children are always my priority.”
According to participants, PCC should be reimbursed because it is preventive care. If they had to
pay, the majority would be willing to pay a fee below 25 euro.

DISCUSSION
Summary of findings
This manuscript presents consumer research to drive socially marketed strategies for delivery of
individual PCC consultations. The most profound finding was the lack of knowledge about the
content and potential benefits of the product. Fertility and psychosocial aspects of parenthood are
components which should be added to PCC. This study points out a key role for health professionals
to promote PCC during moments in routine care with an explainable link to relevance of PCC.
Participants find the community based primary care setting (GP’s and midwives) to be the most
suitable place for PCC. Regarding price, a fee will influence who is reached with the PCC service.

Comparison to the literature

This is not the first study to employ a social marketing approach within the field of PCC; however,
studies define their product differently. Lewis and co-authors define their product as preconception
health and performed a formative inquiry regarding women’s preferences regarding preconception
health.11 Quinn and co-authors confined their product to a single preconception measure:
preconception folic acid supplements. Their intervention approach was a collective campaign.14 To
our knowledge this is the first study in which the product is confined to a specific approach for PCC,
namely individual comprehensive PCC consultations.
To our knowledge there are no studies assessing the effectiveness of social marketing
approaches for preconception care in terms of uptake of services or behavioral change.
Perceptions about preconception care have been assessed in numerous studies. Regarding
the ‘product’, the general misconception of the need and perceived benefits of preparing for
a healthy pregnancy, has been acknowledged as the primary challenge to overcome in the
delivery of preconception care.4,15-17 The need to address fertility and psychosocial aspects of
parenting during PCC is in line with reported low knowledge about fertility (e.g. fertile days) and
timing of parenthood.18 Regarding ‘place’, prior studies underline the preference of women for
GP’s and midwives to be the primary providers of PCC.7,15,19,20 Regarding ‘promotion’, it has been

170
recommended that health care professionals point out PCC in the event of a negative pregnancy
test, when birth control is discussed and in the check-up following delivery of a baby. 15,21,22
This
study supports that the proposed moments are in line with women’s preferences. To our knowledge
there are no studies that assess the effectiveness of pointing out PCC during routine daily care in
terms of promotion of the uptake of PCC.

Strengths and limitations

We believe one of the strengths of this study is that the product is confined to a specific approach:
individual comprehensive preconception care. Firstly, findings over the remaining P’s are valid as
respondents are all talking about the same approach to PCC. Secondly, this way the social marketed
intervention plan is in line with recommendations of the Dutch health board and guidelines of
GP’s and midwives.3,23,24 By taking these points into account, results are close to the situation in
practice, which is important for feasibility of implementation of the approaches which derive from
our findings.
Ideally studies about PCC are performed with a study population that is trying to conceive.
However, these women are not detectable within the general population. Therefore, we employed
a second best approach: women were included if they did not exclude having a pregnancy wish in
the future. This caused the study population to include women throughout various stages of their
reproductive life. We believe our study population to be a representative study sample of planners
and non-planners and nulliparous and multiparous women. We explored patterns regarding
planners/ non-planners; nulliparous/ multiparous and women with prior adverse pregnancy 8
outcomes. However, preferences regarding components of the social marketing plan were not
consistent within these groups, due to the small size of these subgroups. A limitation due to the

towards social marketed preconception care

recruitment in GP and midwifery practices is that results only apply to women that utilize health
care. We recommend effectiveness of approaches that derive from our findings to be evaluated for
subgroups to fine-tune intervention strategies.
This study presents findings in the Netherlands, where individual comprehensive PCC
consultations in primary care are advocated in policy and guidelines. Many countries explore
roles of GP’s and midwives in the delivery of PCC.24,25-30 Findings of this study could be valuable to
such countries or countries with a strongly developed primary care system seeking an individual
approach to preconception care. Furthermore, the methods of consumer research employed in
this study could be illustrative to other countries with other preferred approaches to PCC.

171
CONCLUSION
Preferences of women are largely in line with how PCC is intended to be delivered by primary
care givers. Explicit matters that need rethinking are (1) product: adding fertility matters and
psychosocial aspects of parenthood to the contents of PCC, (2) place: how PCC can be made
accessible to subgroups such as the working population and the low health literate population (3)
price: PCC is currently not reimbursed within basic health insurance whilst a substantial proportion
of women is not willing to pay for individual PCC.
The most profound finding in this study was the low knowledge about contents, benefits
and availability of individual PCC consultations. This emphasizes the importance of promotion.
Participants point out a central role for GP’s and midwives in promoting PCC. They should feel
empowered to promote PCC during the proposed moments. Furthermore, they should point out
PCC regardless of the presence of risk factors; participants prefer to know about availability of PCC
so they can decide whether they want to utilize PCC. However, the low knowledge about PCC and
the fact that midwives generally do not see non-pregnant women provide rationale for a campaign
within the public health sector additional to efforts of PCC providers. This would reach women that
do not visit health care providers and it could sensitize the public to messages about PCC from
health care providers
Our consumer research provides the foundation for a socially marketed programmatic approach
to individual PCC care. An approach needs to be designed in which the identified preferences are
met. The low knowledge and perceived need for PCC entails that there is a need for a continuous
promotion strategy parallel to delivery of PCC. A promotion campaign needs to be developed and
evaluated regarding their comprehensiveness and appeal to different target groups. Feasibility of
meeting women’s preferences needs to be evaluated with PCC providers and policy makers. The
designed program needs to be delivered iteratively, with continuous monitoring and adaption to
specific target audiences.

CONFLICTS OF INTERESTS
None declared.

ACKNOWLEDGEMENTS
We thank the all participants and recruiting practices (General Practice de Jong (Zoetermeer, the
Netherlands), General Practice V&V (the Hague, the Netherlands) and Midwifery practice Groei
(Amsterdam, the Netherlands). We thank Elaine Joziasse- Fitzpatrick for her help in revising, as a
native English speaker.

172
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1. Temel S, van Voorst SF, de Jong-Potjer LC, et al. The Dutch national summit on preconception care: a summary of defini-
tions, evidence and recommendations. Journal of community genetics. 2015;6(1):107-115.
2. Jack BW, Atrash H, Coonrod DV, Moos MK, O’Donnell J, Johnson K. The clinical content of preconception care: an overview
and preparation of this supplement. American journal of obstetrics and gynecology. 2008;199(6 Suppl 2):S266-279.
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2007. publication no. 2007/19E, ISBN 978-90-5549-657-0.
4. van der Pal-de Bruin KM, le Cessie S, Elsinga J, et al. Pre-conception counselling in primary care: prevalence of risk factors
among couples contemplating pregnancy. Paediatric and perinatal epidemiology. 2008;22(3):280-287.
5. van Voorst S, Plasschaert S, de Jong-Potjer L, Steegers E, Denktas S. Current practice of preconception care by primary car-
egivers in the Netherlands. Eur J Contracept Reprod Health Care. 2016;21(3):251-258.
6. van der Zee B, de Beaufort ID, Steegers EA, Denktas S. Perceptions of preconception counselling among women planning a
pregnancy: a qualitative study. Fam Pract. 2013;30(3):341-346.
7. Temel S, Birnie E, Sonneveld HM, et al. Determinants of the intention of preconception care use: lessons from a multi-ethnic
urban population in the Netherlands. International journal of public health. 2013;58(2):295-304.
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ment. first edition ed. Washington, USA: Jossey-Bass; 1995.
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10. Prue CE, Daniel KL. Social marketing: planning before conceiving preconception care. Matern Child Health J. 2006;10(5
Suppl):S79-84.
11. Lewis MA, Mitchell EW, Levis DM, Isenberg K, Kish-Doto J. Couples’ notions about preconception health: implications for
framing social marketing plans. Am J Health Promot. 2013;27(3 Suppl):S20-27.
12. van Voorst SF, Vos AA, de Jong-Potjer LCA, Waelput AJM, Steegers EAP, Denktaş S. Effectiveness of general preconception
care accompanied by a recruitment approach: protocol of a community based cohort study (the Healthy Pregnancy 4 All
study). BMJ Open. Accepted for publication december 2014.
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13. NVivo qualitative data analysis software [computer program]. QSR International Pty Ltd; Version 10, 2012.

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14. Quinn GP, Hauser K, Bell-Ellison BA, Rodriguez NY, Frias JL. Promoting pre-conceptional use of folic acid to Hispanic women:
a social marketing approach. Matern Child Health J. 2006;10(5):403-412.
15. Tuomainen H, Cross-Bardell L, Bhoday M, Qureshi N, Kai J. Opportunities and challenges for enhancing preconception
health in primary care: qualitative study with women from ethnically diverse communities. BMJ Open. 2013;3(7).
16. Hosli EJ, Elsinga J, Buitendijk SE, Assendelft WJ, van der Pal-de Bruin KM. Women’s motives for not participating in precon-
ception counseling: qualitative study. Community genetics. 2008;11(3):166-170.
17. Temel S, Erdem O, Voorham TA, Bonsel GJ, Steegers EA, Denktas S. Knowledge on preconceptional folic acid supplementa-
tion and intention to seek for preconception care among men and women in an urban city: a population-based cross-sec-
tional study. BMC Pregnancy Childbirth. 2015;15:340.
18. Lampic C, Svanberg AS, Karlstrom P, Tyden T. Fertility awareness, intentions concerning childbearing, and attitudes towards
parenthood among female and male academics. Hum Reprod. 2006;21(2):558-564.
19. Mazza D, Chapman A. Improving the uptake of preconception care and periconceptional folate supplementation: what do
women think? BMC Public Health. 2010;10:786.
20. Poppelaars FA, Cornel MC, Ten Kate LP. Current practice and future interest of GPs and prospective parents in pre-concep-
tion care in The Netherlands. Fam Pract. 2004;21(3):307-309.
21. Frishman G. Preconceptional counseling and care: a unique window of opportunity. Medicine and health, Rhode Island.
2003;86(1):16-18.
22. Stern J, Bodin M, Grandahl M, et al. Midwives’ adoption of the reproductive life plan in contraceptive counselling: a mixed
methods study. Hum Reprod. 2015;30(5):1146-1155.
23. de Jong-Potjer L, Beentjes M, Bogchelman M, Jaspar AHJ, Van Asselt KM. The Preconception care guideline by the Dutch
Federation of GP’s 2011; English translation of the Preconception care guideline by GP’s Available at: https://guidelines.

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24. De Jonge AA. Standpunt Preconceptiezorg. Bilthoven: Koninklijke Organisatie van Verloskundigen (KNOV);2005.
25. Shawe J, Delbaere I, Ekstrand M, et al. Preconception care policy, guidelines, recommendations and services across six Eu-
ropean countries: Belgium (Flanders), Denmark, Italy, the Netherlands, Sweden and the United Kingdom. Eur J Contracept
Reprod Health Care. 2014:1-11.
26. Kitamura K, Fetters MD, Ban N. Preconception care by family physicians and general practitioners in Japan. BMC Fam Pract.
2005;6:31.
27. Heyes T, Long S, Mathers N. Preconception care - Practice and beliefs of primary care workers. Family practice. 2004(21):22-
27.
28. Callegari LS, Ma EW, Schwarz EB. Preconception Care and Reproductive Planning in Primary Care. The Medical clinics of
North America. 2015;99(3):663-682.
29. Best Start Resource Centre. Preconception Health: Physician practices in Ontario. Toronto, Ontario, Canada2009.
30. Chuang CH, Hwang SW, McCall-Hosenfeld JS, Rosenwasser L, Hillemeier MM, Weisman CS. Primary care physicians’ per-
ceptions of barriers to preventive reproductive health care in rural communities. Perspectives on sexual and reproductive
health. 2012;44(2):78-83.

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8

towards social marketed preconception care

175
SUPPLEMENTARY FILE: Semi-structured item list.

Brief introduction to the study:

“Purpose of this interview study is to learn how individual preconception care consultations could
best be offered, according to women.”
Check if participant complies with inclusion criteria.
“Therefore we ask women between 18 and 41 years old, that wish to have a child in the near
future, later in life or are sure not to have a completed family yet, to participate. Does this apply to
you?”
The participant has filled in the base-line questionnaire.

Product

A. Preknowledge Firstly assess what people know about the product.

1. What is a PCC consultation? (Preknowledge)

2. What do you think is the goal of PCC consultation? (Preknowledge)
3. Would you utilize the possibility of a PCC consultation if you had a child wish?
(Preknowledge)
4. Why would or wouldn’t you make use of a PCC consultation? (Preknowledge)

B. Presentation with our product

Assure that from now on the interview is about the product.
o I’ll now explain our vision on preconception care, so we are sure we are talking about the
same thing for the rest of the interview. It is possible to visit a preconception care service if
you are thinking about becoming pregnant. During the consultation you can ask questions
about fertility, getting pregnant, your health and you will receive advice about what you can do
to be optimally prepared for a healthy pregnancy. In the first months the baby is very
vulnerable: for example important organs such as heart and lungs are formed. During the PCC
consultation advice is given about your personal health (e.g. regarding medication use
because they can give deformities to the fetus, or regarding hereditary diseases) and general
advice is given (for example folic acid supplementation). Preconception care consultations are
for everybody in any stage of pregnancy contemplation. Furthermore it includes a thorough
risk analysis after which individual advice is provided.”
5. After this explanation, would you utilize the possibility of a Preconception Care Consultation if
you had a child wish? Why or why not? (Utilization of product)
6. What would be important reasons for you to utilize an individual preconception care
consultation service? (Utilization of product)
7. What would be barriers to utilize an individual preconception care consultation service?
(Utilization of product)
8. What would you like to address during a preconception care consultation regarding your
pregnancy wish? (Content of consultation)?
9. Who should deliver individual PCC to you? (Delivery by who)
10. What makes this person the most appropriate provider of individual PCC to you? (Delivery by
who)
11. Should individual PCC always be delivered by a health care professional? Yes/ no and why?
(Alternatives)
12. Which alternatives for delivery by a health care professional would you find suitable?
(Alternatives)
13. What is your opinion about digital media to provide you with preconception information (for
example an app or website)? (Alternatives)
14. Could this replace an individual consultation with a health care professional? (Alternatives)

176
 Promotion

15. How would you prefer to be informed about the possibility to visit a health care professional for
a PCC consultation? (How)
16. Which moments in daily life would you find suitable to be informed about a PCC consultation?
(When)
17. During which phase in your life would you like to be appointed the possibility to visit a health
care professional for a PCC consultation? (When)
18. Which health care professional would you prefer to inform you about the opportunity to visit a
health care professional for a PCC consultation (by this professional or by a different
professional)? (Who)
19. During the next few questions I will mention moments during which you might visit a health
care professionals. How suitable are these moments for health care professionals to point out
the possibility of a preconception care consultation? Please tell us why moments are or aren’t
suitable and provide a grade between 1 -10 for their suitability (1 being absolutely unsuitable
and 10 being very suitable). (When)
a. When I mention my pregnancy wish
b. During regular follow-up of a chronic disease (e.g. check-up for Diabetes)
c. When hereditary diseases are discussed
d. When a medication is prescribed to me, which hasn’t been prescribed to me before
e. When contraception is discussed
f. When a pregnancy test is done and turns out to be negative
g. When I have questions about fertility
h. In the care after a miscarriage 8
i. During a consultation after I had a baby
j. When there are/ were health problems with my baby/child

towards social marketed preconception care

20. a. What do you think about a more anonymous approach to inform you about the possibility of
an individual PCC consultation?
b. Would you prefer an anonymous approach or an approach by a professional or somebody
else? (How)
21. Would you only prefer to be informed when you have risks or always (regardless of your
health)? (When)

Place

22. What do you find important regarding the place where the individual PCC consultations are
provided? (Where)
23. Where would you like the PCC consultation to be provided? (Where)
24. Which moment would you find suitable for a PCC consultation to take place? (When)
25. What is important regarding the moment at which PCC is offered? (When)

Price

26. Would you be willing to pay for a PCC consultation? Why or why not? (Acceptance)
27. How much would you maximally be willing to pay for a PCC consultation? (Amount)

Do you have any remaining remarks or questions?

We thank you for your participation.

177
IMPLEMENTATION OF A
COMMUNITY-BASED PEER HEALTH
EDUCATION STRATEGY FOR
PRECONCEPTION CARE
S.F. van Voorst, V.L.N. Schölmerich, Carissah J.C. Stewarts,
D.W. van Veen, E.A.P. Steegers, S. Denktaş.
Manuscript submitted
ABSTRACT
Objective: To evaluate the implementation of peer education as a strategy to a) reach
underserved women of reproductive age with preconception peer education and to b) refer
them to preconception care (PCC) consultations in primary care (the Healthy Pregnancy 4 All –
Preconception care substudy) in fourteen Dutch municipalities.
Methods: Process evaluation was performed according to Saunders Guideline for Process
Evaluation. Implementation criteria were applied to assess the extent to which process measures
regarding dose delivered, dose received, fidelity and outreach, were achieved.
Results: The intervention was adopted in seven out of ten municipalities. Overall implementation
rates for items regarding dose delivered was 100% and 81% regarding dose received.
Implementation fidelity was 62% and led to low outreach amongst the target population (49%).
The strategy led to uptake of individual PCC by one woman. We identified several explanations for
the insufficient implementation.
Conclusion: The implementation strategy is feasible, yet needs improvement before conclusions
can be drawn regarding the effectiveness of the strategy.
Practical implications: Improved strategy should invest in 1) developing working relationships
between peer educators and PCC providers; 2) consensus amongst stakeholders regarding the
target group and 3) developing recruitment strategies for peer education sessions.

180
INTRODUCTION
Peer health education, or ‘the teaching and sharing of information, values and behaviours, between
individuals with similar characteristics’, is a popular approach in the field of health promotion.1
This form of education – referred to as ‘peer education’ from now on, has been successfully
employed to increase women’s knowledge about preconception health.2-4 However, education
alone is not sufficient to reduce preconception risks. Peer education provides general information
to participants, while the majority of couples have one or more risk factors for which a visit to a
general practitioner (GP) is indicated.5, 6
Preconception care (PCC) aims to optimize the health of (future) parents and their babies
by reducing risks amongst prospective parents before pregnancy. The contents of care ranges
from interventions which are applicable to all future parents (e.g. folic acid supplementation) to
interventions applicable to specific risks amongst parents (e.g. optimizing glycaemic control in case
of diabetes).7, 8 A study estimated that up to 35% of perinatal complications could be prevented by
addressing risk factors with PCC.9
Recent studies suggests that peer education is an effective strategy for reaching participants
that are from disadvantaged neighbourhoods and typically “underserved” by PCC services.4, 10 The
intervention described in this article aims to a) reach underserved women of reproductive age

BACKGROUND
The intervention
During this peer education intervention, trained peer health educators (called peer educators
onwards) delivered education sessions about preconception health to women from disadvantaged 9
neighborhoods. The aim of the sessions was to cover behavioral risks (e.g. intake of folic acid

Implementation of peer health education

supplements or illicit substance use) and medical risks (e.g chronic diseases, prior obstetric
complications) associated with adverse pregnancy outcomes. Peer educators were to refer
participants to a preconception consultation services at GP’s and midwifery practices that
participated in the Healthy Pregnancy 4 All program (HP4All). The sessions aimed to be interactive
and informal, and to take place within local and familiar settings (e.g. a community center or
participants’ home).

Setting and context of the intervention

The peer education approach presented in this article is part of the preconception care sub-study
within the Healthy Pregnancy 4 All program (HP4All).11 This sub-study is a cohort study conducted
between 2011 and 2014. Its aim was to improve perinatal health in disadvantaged neighborhoods
of 14 municipalities with perinatal mortality rates that exceeded the national average of 10:1000
births.12, 13 The sub-study had two organizational goals: (1) delivery of PCC consultations in primary
care (by GP’s and midwives in the community) and (2) develop a recruitment strategy to target
women aged 18 – 41 to utilize these PCC consultations. The peer-education intervention described
in this article was one of these recruitment strategies.

181
 Similar to other countries, PCC in Dutch primary care is fairly new and – before the intervention
- only offered at a small scale.14 Therefore, we set up dedicated PCC centers, where providers were
trained and assisted in their delivery of PCC. The HP4All program achieved that a median of 88%
(50-100%) of the midwife practices in the municipalities and a median of 17% (12-75%) of the GP
practices in the municipalities participated as PCC delivery centers. Preconception peer educators
were not present in the municipalities before.

THE IMPLEMENTATION STRATEGY

Figure 1 depicts our implementation strategy, or the chain of events that should eventually lead to
improved preconception health.
Inputs consist of tasks performed by HP4All staff to initiate the intervention. HP4All staff
consisted of staff at the national level and a program manager for each municipality. Two
members of the HP4All staff were responsible for the roll out and the evaluation of the peer
education approach. The HP4All program aimed to achieve adoption of the peer education
strategy by bringing the program on the municipal health agenda via the local program manager.
Local program managers were either attached to the municipal health service or the municipal
department of public health. They were responsible for the local roll out of the intervention. Local
program managers were seen as crucial to provide adaptation to the local setting, access to existing
networks with health care professionals and knowledge of ways to reach the target group.

Immediate impacts
Once the municipality agreed to adopt peer education, the local program manager was
responsible for the recruitment of the peer educators. The selection criteria for the recruitment
of peer educators were (1) completion of at least upper secondary level education, (2) adequate

FIGURE 1: The implementation approach for peer health education intervention.

Organizational
Inputs Immediate impacts Short term impacts Health outcomes
outcomes

Individual behavioural
Initiation by HP4All staff Adoption by municipality Implementation
outcomes

Introducing the intervention in · Educators with the Recruitment of women

the HP4All municipality Selection of educators desired profile and a for peer education
certificate for delivery sessions
of PCC are available. Change of
· All certified deliver preconception health
Providing the training for the Training and certification peer education Delivery of peer behaviours
program of educators · Peer education is only education sessions
delivery by certified
educators Uptake of PCC
Materials, reimbursement · Timing is adequate, services
and support for the Referral to HP4ll’s PCC
so sufficient time is services
organization of education left to recruit towards
sessions PCC centers
Process
evaluation Dose delivered Dose received Fidelity Outreach
measures

182
proficiency of the Dutch language and preferably of a second language, (3) knowledge and access
to the hard to reach groups in the community, (4) cultural sensitivity and (5) access to organizations
through which participants for peer education sessions could be reached (e.g. a maternity care
organization). Local program managers were responsible for the allocation of budget for actual
delivery of peer education sessions and for supplying educational materials suitable for the local
setting.
Development and delivery of the training was delegated to one training bureau that facilitated
training in each municipality. The training was designed as a post-secondary course. It consisted
of eight modules ranging from practical skills (communication, presenting, organization of
sessions) to theory (general health and common diseases, reproductive health, preconception
health, parenting and perinatal health care in the Netherlands).4 The educators-in-training were
assessed with assignments and a practical exam and rewarded with a certification. The training was
scheduled to take a maximum of six months.

Short term outcomes and organizational outcomes

The prior mentioned efforts were expected to result in certified educators that were ready to deliver
peer health sessions as soon as the GP’s and midwives were ready to receive referred participants
for PCC consultations. Peer educators and program managers were tasked to develop a recruitment
approach based on their knowledge of the local target group and the available networks to reach
them.
The efforts of trained peer educators were expected to result in outreach. We define outreach as:
1) recruitment of women of reproductive age to participate in peer education sessions 2) delivery
of peer education sessions to this target group and 3) referral to HP4All PCC consultation services. 9
Health outcomes

Implementation of peer health education

Peer education sessions were expected to result in the uptake of HP4All’s PCC consultations. Results
of the effectiveness of PCC consultations services and the recruitment strategy will be described
elsewhere (see van Voorst et al 2015 for the study protocol).11 with preconception peer education
sessions and to b) have peer health educators refer these women to local preconception care
consultation services, therefore promoting their uptake of PCC.
This article provides the process evaluation of the implementation strategy to in seven
municipalities in the Netherlands. The performed process evaluation is important for formative
purposes to understand the extent to which an evaluation of the effectiveness of the intervention
is warranted, as well as for summative purposes to improve implementation strategies of future
interventions. To our knowledge, this is the first study reporting a process evaluation for peer
education for preconception health. Likewise, it is the first study outlining an approach to using
peer education for referral to local health care services.

183
 TABLE 1: Process evaluation items, criteria for implementation and results of process evaluation per municipality.

Implementation Met implemen-

criteria per munic- tation criterium
Process evaluation measures Data Source Rating Scale / Coding ipality A B C D E F G **
Dose delivered
1 Did the HP4All team discuss the peer education Logbook 1 = yes (1/1) 1 1 1 1 1 1 1
strategy with the municipal program manager? 0 = no (0/1)
2 Did the HP4All staff provide the training material? Logbook 1 = yes (1/1) 1 1 1 1 1 1 1
Score = 3 (100%) 7/7
0 = no (0/1)
3 Was funding delivered to execute the planned Logbook 1 = yes (1/1) 1 1 1 1 1 1 1
training program? 0 = no (0/1)
Dose received
4 Was there an agreement between to adopt the Logbook 1 = yes (1/1) 1 1 1 1 1 1 1
peer education strategy? 0 = no (0/1)
5 How many peer educators were selected for the Logbook 3=≥4 3 2 3 3 3 2 3
training? 2 = 2 to 4
1=1

184
0=0
6 What was the proportion of peer educators that Logbook 4 = 80 - 100% 2 4 4 2 4 2 2
completed the training? 3 = 60 - 80%
2 = 40 - 60%
1= 20 - 40%
0 = >20 %
7 Did HP4All provide material for the peer educa- Logbook 1 = yes (1/1) 1 1 1 1 1 1 1
tion sessions to the municipalities? End of program interview (program manager 0 = no (0/1)
and peer educators Score ≥8 (≥60%) 7/7

8 Did the municipal program manager ensure peer Logbook 1 = yes (1/1) 1 1 1 1 1 1 1
educators with material for the sessions? End of program interview (program manager 0 = no (0/1)
and peer educators
9 Did the municipal program ensure reimbursement Logbook 1 = yes (1/1) 1 1 1 1 1 1 1
of costs of the sessions? End of program interview (program manager 0 = no (0/1)
and peer educators
10 Did the municipal program manager provide Logbook 1 = yes (1/1) 1 1 1 0 1 0 1
support to peer educators in developing the End of program interview (program manager 0 = no (0/1)
recruitment strategy for the target group? and peer educators
11 Did municipal program managers bring peer Logbook 1 = yes (1/1) 0 0 1 0 1 0 0
educators in contact with PCC health centers? End of program interview (program manager 0 = no (0/1)
and peer educators
 Fidelity
12 Were peer educators ready to deliver group Logbook 1 = yes (1/1) 0 1 0 0 1 0 1
sessions as of the moment that PCC services were End of program interview (program manager 0 = no (0/1)
available? and PHE)
13 Was the intended course executed as intended? Logbook 1 = yes (1/1) 0 0 0 0 1 0 0
End of program interview (program manager 0 = no (0/1)
and peer educators
14 Did the candidates have the intended profiles? Logbook 1 = yes (1/1) Score ≥4 (≥60%) 1 1 1 1 1 1 1 4/7
0 = no (0/1)
15 Were all the sessions provided by peer educators Logbook 1 = yes (1/1) 1 1 1 1 1 0 0
certified by the program? 0 = no (0/1)
16 Which proportion of the certified peer educators Logbook 2 = 100% 2 2 2 1 2 0 0
organized sessions? 1 = 50 -100%
0 = < 50
Outreach
17 How many peer education sessions were there Peer education registration forms 4 = ≥ 15 2 1 3 2 4 0 4
organized? 3 = 10 to 15
2 = 5 to 10
1 = 1 to 5

185
0=0
18 How many participants attended the peer educa- Peer education registration forms 4 = ≥ 150 3 1 2 1 4 0 4
tion sessions? 3 = 100 to 150
2 = 50 to 100 Score ≥6 (≥60%) 4/7
1 = 10 to 50
0=0
19 To what extent did the educators refer to the End of program interview (program manager 3= generally always and 1 2 2 0 2 0 1
HP4All PCC services? and peer educators when women have risk
factors; 2= always; 1= dur-
ing some of the sessions;
0= did no

Implementation of peer health education

9
METHODS
The steps of our process evaluation are described below and were based on Saunders Guideline for
process evaluation of health promotion strategies.15

Development of process evaluation measures

We drafted a chain of events model, as displayed in Figure 1, to break down each event into
single actions that have to happen for the given event to take place. We then formulated process
evaluation measures and scoring criteria for each action, as shown in Table I. For example, one
of the actions was that only certified peer educators should provide peer education. Adherence
to this principle was evaluated with process measure 15: “Were all the sessions provided by peer
educators certified by the program?” If our data indicated that all sessions were provided by a
certified educator, one point was given, if not, zero points were given.
In a next step, we clustered our process evaluation measures into implementation
components: dose delivered, dose received, fidelity, and “outreach”. Dose delivered (labelled
‘input’ in figure 1) describes to what extent the HP4All team delivered the specified actions to
initiate the implementation strategy. Dose received (‘immediate impacts’) explains to what extent
municipalities responded to the dose delivered. Fidelity (‘short term impacts’) explains to what
extent the intervention was implemented as originally planned. In turn, this was expected to result
in outreach (‘organizational outcomes’). We defined outreach as the scale at which the population
was reached by peer education and referred to PCC consultations. Table I presents the process
measures and scoring criteria per implementation component.

Data collection
We collected implementation data from the start of the intervention (September 2014) until its
completion (July 2014). Data collection consisted of:
• Logs and program administration files about the training and selection of peer educators. This
data was collected by the national HP4All staff.
• End of program interviews held with one purposively selected peer educator from each
municipality and all program managers. During these interviews we went through the process
evaluation measures of Table I and cross-checked the data from our other sources with the
respondent’s accounts. Additionally, we asked respondents about constraints and facilitating
elements in the implementation process to identify factors that may have influenced the
implementation. These answers were transcribed and organized per topic in a spread-sheet.
• Questionnaires for educators were filled in by educators after each education session

throughout the entire intervention. This included data about the location and date of the given
session, the number of participants and how the participants were recruited.
• Questionnaires for participants were handed out to all participants of the group sessions.
These questionnaires collected data on participants’ characteristics, such as age, education and
whether or not they were planning to conceive.
• The HP4All database of the PCC sub-study: All women who applied for preconception services

186
 participating in the HP4All study were registered in the HP4All database as of September 2011
– December 2014.11 The participating practices registered whether the women were referred by
a peer educator.

Data analysis and applying criteria to assess levels of implementation

Triangulation of the collected data was performed to score the process evaluation measures in
Table I. The first author (SVV) discussed the attributed scores with DVV, and in case of disagreement,
a third scorer (SD) was approached for consensus. We organized data into tables and calculated
sum scores. The overall aim was to achieve adoption of PHE in at least six of the ten participating
municipalities.
We set the implementation criteria at 100% for dose delivered as these items concerned our
own activities and at 60% for the other process evaluation components (dose received, fidelity
and outreach). This means that if 100% of the maximum points within dose delivered, and 60%
within the other implementation components were achieved, we regarded the component to be
implemented sufficiently. Our decision to apply a 60% criterion was based on a review by Durlac
and Dupre.16 They state that program implementation rates above 80% are rare and that a rate of
60% tends to produce an effective program.
Our process evaluation showed which steps were implemented insufficiently and helped
understand why steps did or did not go as planned. Based on these findings, we developed an
improved implementation strategy (supplementary file 1).

RESULTS
9
Adoption

Implementation of peer health education

Pear education was adopted by seven municipalities (the Hague, Enschede, Groningen, Nijmegen,
Schiedam, Tilburg and Utrecht). Reasons for other municipalities not to adopt the intervention
were lack of governmental funding and concerns regarding the feasibility of implementation
within the timeframe of three years. In this study we evaluate the implementation process of the
seven municipalities that adopted the strategy.

Implementation
Figure 2 shows how well the municipalities scored per process evaluation measure. These results
were computed by aggregating the scores of the seven municipalities per process measure. Table I
provides the scores per municipality.
We will discuss the highlights of our process evaluation based on items in Figure 2 and the
scores in Table I. We provide additional data extracted from the data sources specified in the
methods section to add contextual information to our evaluation.

187
 DOSE DELIVERED*
FIGURE 2: A

1. advocation of peer education strategy in municipality

2. provision of training material
3. provision of funding for training
DOSE RECEIVED*
4. municipal adoption of the peer health education intervention
5. sufficient numbers of educators recruited
6. sufficient completion of the training by selected candidates
7. provision of material to municipality
8. provision of material to peer educators
9. provision of reimbursement for education sessions
10. support of educators to recruit groups

188
11. coordination to set up networks between peer educators and HP4All PCC centers
FIDELITY AND COMPLETENESS*
12. timing of availability of peer education sessions
13. completion of course as intended
14. profiles of candidates as intended
intervention Numbers correspond with process measures in Table I.

15. education sessions were exclusively delivered by certified educators

16. all certified educators delivered education sessions
ORGANIZATIONAL OUTCOME*
17. number of PHE sessions organized
18. numbers of women reached with group sessions
19. referral was made to PCC services

0% 20% 40% 60% 80% 100%

computed by aggregating the scores for the seven municipalities that adopted the peer education
 ssessment of the overall implementation of the peer health education strategy. These scores were
Dosage
All participating municipalities reached 100% for dose delivered. The HP4All program succeeded
in advocating the strategy in the municipality (measure 1). This was enabled by the partnership
between the national HP4All staff and the municipal program manager. Furthermore, HP4All
succeeded in providing budget and material for the training (measure 2, 3). The training material
was largely adapted from a previous Dutch peer education health program that included
preconception health.4
Overall, the municipalities scored well on dose received (81%). The municipalities recruited
the desired number of peer educators (measure 5) and the drop-out rate of the peer education
training was within the anticipated range (measure 6; drop-out rate of 15% amongst 41 selected
candidates). Low satisfaction with the training was the main reason for drop-out. Peer educator
candidates indicated that they experienced low satisfaction as there was not enough practice in
actual delivery of education sessions and the training required much more time than anticipated.
Moreover, candidates who were already active as peer educators in other health topics felt that the
training was redundant.
There were no problems with the distribution of material (measure 7, 8) and the financing of
the education sessions (measure 9). Municipalities adhered to the recommended material. Peer
educators and program managers indicated that the materials were versatile and could be used in
different ways to meet the different knowledge levels of the participants. Moreover, municipalities
provided adequate support in the recruitment of participants in five of the seven municipalities
(measure 10). Educators from two municipalities had to rely on their own network to the target
population which they perceived to be low.
Within the dose received component, one item that was not achieved sufficiently: peer 9
educators and participating PCC centers in the municipalities were not brought into contact

Implementation of peer health education

with each other by program managers in five of the seven municipalities (measure 11). This is
problematic, as this step was seen as essential so that educators could form a dedicated network
for referral. In end of program interviews, program managers stated that they did not connect
educators with PCC providers due to time restraints. They were more focused on the completion of
the training, setting up recruitment strategies and the organization of the peer education sessions.
The program managers indicated that developing relationships with primary caregivers would
have needed more time and multiple contacts rather than a single effort just to get acquainted.
They foresaw that there was not sufficient time for this within the program. With the exception of
two municipalities, peer educators did not approach the caregivers that delivered PCC within the
program themselves. They joined existing meetings of GP’s and/ or midwives and sent letters to
GP’s and midwives. Despite enthusiasm of these primary caregivers, these efforts did not result in a
working relationship.

189
Fidelity
The overall score for fidelity, or the extent to which the intervention was implemented by program
members as planned, was 62%. Lower scores on fidelity in all municipalities were due to the
adjustment of the training (measure 13) and the late initiation of peer education sessions (measure
12).
The training for peer educators was not delivered as intended (measure 13). Program managers
requested to shorten the training as many educators had ample pre-training knowledge. The
course was restricted to the contents of preconception health and only briefly addressed practical
aspects of recruiting participants and the delivery of peer education sessions (i.e. presentation
training). Retrospectively, some peer educators reported that more practical training would have
been better.
Fidelity was mostly compromised because peer education sessions were delivered later than
planned (measure 12). It was planned that peer educators would deliver peer education as soon
as preconception consultation services were available at participating GP and midwifery practices.
Program managers and educators explained that late delivery of peer education sessions was due
to a late completion of the training and that the strategy did not provide enough time to develop
strategies to recruit women to participate in the educational sessions. To resolve lack of time to
recruit women, PCC was integrated into existing events in the community (e.g. coffee sessions
amongst parents at schools or information meetings about other health conditions) rather than
setting up specific recruitment strategies for preconception peer education. Our analysis provided
that most participants (46%) were reached because the peer education sessions were integrated
into a group event for which participants were already recruited. This approach was applied in
three municipalities. Furthermore, fidelity was reduced in three municipalities, because educators
provided peer education whilst they were not certified or because certified educators did not come
around to provision of peer education time wise.

Outreach
The overall score for outreach of the peer education strategy was insufficient (50%). Although
the overall number of organized peer education sessions was sufficient (86%, item 17), the extent
to which the desired of participants was reached was insufficient (50%; measure 18). A total of
1796 participants attended the group sessions, but many sessions only hosted a small number of
participants (<8).
Table II shows the characteristics of the participants that attended peer education sessions. The
participants were mainly non-Western ethnic minorities, predominantly first generation immigrants,
low to intermediately educated women with low Dutch language proficiency and low knowledge
about PCC. These characteristics are typical for people from disadvantaged neighbourhoods, who
are typically underserved by PCC. Interestingly, 43% of the participants were beyond the targeted
age range (18 up to and including 41 years). Only nine percent of the participants intended to
become pregnant. The fact that peer education was often integrated into existing group events
explains the lack of adherence to the target population and the low number of participants that

190
wished to conceive. Additionally, program managers stated that the developed recruitment
strategy was deliberately not restricted to women of reproductive age. They felt that improving
women’s health literacy and motivation to attend PCC would require talking about preconception
health with teenagers but also with the social network of prospective mothers. Furthermore,
addressing a broader age range about reproductive health was their primary aim rather than the
short term aim of promoting the potential uptake of PCC, within the HP4All program.
Lastly, the extent to which educators referred women to PCC services was insufficient
(adherence to the concept of referral was 38%; measure 19). Peer educators in three municipalities
reported always mentioning the availability of preconception services at GP’s and midwifery
practices. This was only done occasionally in two municipalities, and was never done in the two
remaining municipalities. The educators mentioned the following reasons for not referring to
preconception consultations within the HP4All program were (1) thinking that a participants’
risk factor required referral to the women’s GP which happened not participate as a PCC delivery
center within HP4ALl, (2) sessions were sometimes delivered outside of areas with participating
PCC centers; so people were referred to their own caregiver (who did not participate in the HP4All
program) (3) not knowing to which GP and midwifery practices they could refer women to.

Health Outcome
Within the course of the project one of 587 applications for a PCC consultation at the GP and
midwife practices was registered.

DISCUSSION
9
This study provides a new approach to integrate different preconception approaches: a classic

Implementation of peer health education

approach from the public health care field (peer education) and one within the primary care setting
(individual PCC consultations). Our evaluation shows that seven out of ten municipalities adopted
the intervention strategy. The implementation strategy resulted in 147 peer education sessions,
with a total of 1796 participants. Overall dosage was sufficient; fidelity was marginally sufficient
(62%) and outreach of the program was insufficient (overall score 49.4%). Four of the seven
municipalities satisfied our implementation criteria. However, only one of the 1796 participants of
the peer education sessions visited a PCC service of the HP4All program, although it is possible that
more participants made use of non HP4All PCC services.
Our process evaluation helped us to identify explanations for low outreach: 1) deviation
from the intended target group (women of reproductive age) 2) insufficient dedication and time
allocated to the development of the referral network between educators and PCC caregivers 3)
referral was not performed consistently or beyond the desired network of PCC providers within the
project.
Peer education has been implemented in the field of PCC before.2-4, 10, 17, 18 These studies were
mostly conducted amongst underserved populations. These studies show that peer education
can reach underserved women. However, none of the studies referred women to individual PCC

191
 TABLE 2: Background characteristics of participants of the preconception peer education sessions per municipality..

Municipality A B C D E F Total
N= 20 (resp. rate = N= 37 (resp. rate = N= 45 (resp. rate = N= 13 (resp. rate = N= 856 (resp. rate N= 156 (resp. rate N=1127
18.5%) 90.2%) 81.8%) 26.0%) = 100%)9 = 22.8%) resp. rate 63%
Age Years (median (range)) 38 (26-71) 48 (32-68) 30 (19-59) 44 (28-67) 27 (13-76) 40 (19-66) 31 (13-76)
Ethnicity1 Native Dutch: 0 (0) 0 (0) 30 (66.7) 5 (38.5) 174 (20.3) 4 (2.6) 213 (18.9)
Surinamese 2 (10.0) 0 (0) 0 (0) 0 (0) 50 (5.9) 1 (0.6) 53 (4.0)
Antillean/Aruban: 0 (0) 0 (0) 1 (2.2) 2 (15.4) 36 (4.2) 0 (0) 39 (3.5)
Indonesian/Moluccan: 0 (0) 0 (0) 1 (2.2) 0 (0) 15 (1.8) 0 (0) 16 (1.4)
Turkish 5 (25.0) 36 (97.3) 0 (0) 4 (30.8) 233 (27.2) 76 (48.7) 354 (31.4)
Kurdish 3 (15.0) 0 (0) 0 (0) 0 (0) 6 (0.7) 0 (0) 9 (0.8)
Moroccan 7 (35.0) 0 (0) 8 (17.8) 1 (7.7) 134 (15.7) 69 (44.2) 219 (19.4)
Polish 0 (0) 0 (0) 0 (0) 0 (0) 23 (2.7) 0 (0) 23 (2.0)
Other 3 (15.0) 0 (0) 5 (11.1) 1 (7.7) 171 (20.0) 3 (1.9) 183 (16.2)
Immigrant status2 First generation 20 (100) 34 (91.9) 9 (20.0) 9 (69.2) 413 (48.2) 124(79.5) 609(54.0)

192
Second generation 0 (0) 2 (5.4) 9 (20.0) 1 (7.7) 273 (31.9) 26 (16.7) 311 (27.5)

Native Dutch: 0 (0) 0 (0) 27 (60.0) 3 (23.1) 139 (16.2) 4 (2.5) 173 (15.4)
Civil status Married or living together 17 (85,0) 31 (83,8) 24 (53,3) 8 (69,2) 77 (72.6) 118 (75,5) 276 (63)
In a relationship, not living together 0 (0) 0 (0) 6 (13.3) 0 (0) 8 (7.5) 4 (2.6) 18 (4.8)
Not in a relationship 3 (15.0) 6 (16.2) 15 (33.3) 4 (30.8) 18 (17.0) 29 (18.6) 75 (19.9)
Residence3 Non-deprived neighbourhood 1 (5.0) 29 (78.4) 42 (93.3) 9 (69.1) 557 (65.1) 56 (35.9) 694 (61.6)
Deprived neighbourhood 18 (90.0) 6 (16.2) 1 (2.2) 3 (23.1) 293 (34.2) 93 (59.6) 414 (36.7)
Educational attainment Low 13 (65) 27 (73) 12 (73) 5 (38.5) 481 (56.2) 68 (60.5) 625 (53.8)
level4
Intermediate 3 (15.0) 6 (16.2) 22 (48.9) 5 (38.5) 191 (22.3) 42 (26.9) 269 (23.9)
High 1 (5.0) 0 (0.0) 8 (17.7) 2 (15.4) 110 (12.8) 21 (13.5) 142 (12.6)
Other 2 (10.0) 1 (2.7) 2 (4.4) 0 (0) 26 (3.0) 4 (2.6) 35 (3.1)
Occupational status5 Unemployment 14 (93.3) 29 (80.6) 27 (62.8) 7 (53.8) 24 (27.6) 106 (72.1)
 Language Proficiency6 Excellent 1 (5.0) 5 (13.5) 36 (80.0) 9 (69.2) 43 (49.4) 51 (32.7) 145 (40.5)
Sufficient 7(35.0) 15 (40.5) 6 (13.3) 2 (15.4) 23 (26.4) 42 (26.9) 95 (26.5)
Moderate 5 (25.0) 6 (16.2) 2 (4.4) 0 (0) 7 (8.0) 21 (13.5) 41 (11.5)
Insufficient 6 (30.0) 10 (27.0) 36 (80.0) 9 (69.2) 43 (49.4) 51 (32.7) 145 (40.5)
Knowledge about PCC7 Mean score, (SD) 4.94 (3.33) 4.14 (1.86) 7.55 (2.00) 7.10 (2.80) 4.63 (2.49) 5.40(2.49) 5.46 (2.64)

Pregnancy intention8 Yes 6 (30.0) 1 (2.7) 6 (13.3) 0 (0) 6 (6.9) 14 (9.0) 33 (9.2)

- These numbers reflect data from the respondents that agreed to fill in the questionnaire (overall response rate 67.7%)
- Numbers reflect number of participants (N) unless specified differently.
- Percentages do not always count up to 100% due to missing data or rounding.
1. Ethnicity is defined as the social or cultural group that the participant considered themselves to be part of.
2. First generation immigrant is defined as somebody that is not born in the Netherlands; a second generation immigrant is defined as somebody that is born in the Netherlands
but either of her parents was born in a foreign country.
3. Classification of living in deprived neighbourhood was performed with zip-codes according to classification of the Central Bureau of Statistics (CBS) in the Netherlands in 2008.
4. Educational attainment level was defined as the highest completed educational level classified according to the International Standard Classification of Education (ISCED) i.e.
low (level 0-2: early childhood; primary education; lower secondary education); intermediate (level 3-5: upper secondary; post-secondary; short cycle tertiary); and high (level
6-8: bachelor; master; doctoral) Unesco institute for statitstics 2014.

193
5. Employment was defined as having a paid job.
6. Language proficiency was self rated and based on an average score for comprehension, reading, writing and speaking.
7. Knowledge about PCC was assessed with 10 questions concerning preconception health, each correct answer is rewarded with 1 point resulting in a maximum score of 10
points; these questions were assessed prior to the peer education session.
8. Pregnancy intention was assessed by asking whether or not women were currently trying to conceive or not.
9. Number of participants was not registered; however PHE reported that all participants filled in the questionnaire. Therefore the response rate is assumed to be approximately
100%.

Implementation of peer health education

9
consultations after they attended peer education sessions.
This article is an illustration of how effectiveness data alone would provide an incomplete
picture of outcomes.19 An effectiveness study would have indicated that peer education is not
effective to improve uptake of individual PCC. With a process evaluation, however, we can show
that potential lack of effectiveness might not be because the program does not work, but because
it was not implemented as intended. Lack of implementation evaluation could result in a “type III”
error, where a program is rejected while it was not implemented as intended.20
Potential limitations of our study are inherent to our program monitoring strategy and the
data sources. The monitoring mostly relied on data that was analyzed or collected at the end of the
program. This was partly due to time constraints.11 Interim evaluations could have flagged barriers
to implementation at an earlier stage. Regarding data sources, one peer educator per municipality
was asked to participate in the end of program interviews and the response rate of the participant
questionnaires varied per municipality between 18.5% and 100%. This could give rise to selective
process evaluation data. Lastly, the way uptake of PCC services was registered might have led to an
underestimate of the actual number of PCC consultations after referral by peer educators. Firstly
because we experienced that PCC consultations were not always registered. Secondly, how women
had heard of the PCC service was not registered amongst 10% of the admissions. Furthermore, the
low percentage of participating GP’s (median 17% (12-75%) might have played a role in referral
to non-participating GP’s. We were unable to collect data regarding referrals for PCC from these
practices.

CONCLUSION
Peer health education can be implemented in community-based health care. This study showed
the potential of peer health education to reach underserved women, however referral to PCC
consultations in primary care was performed inadequately. Therefore, an evaluation of the
effectiveness of the referral strategy on uptake of PCC consultations is not warranted yet. There
is however sufficient ground to adjust the implementation strategy for the purpose of improving
future implementation of (similar) interventions.

PRACTICE IMPLICATIONS
• We recommend introducing a municipal field coordinator dedicated to 1) making and
maintaining contact with PCC providers in primary care, 2) designing and managing recruitment
strategies to recruit the target group for the peer education sessions and 3) monitoring the
skills and training levels of peer educators.
• As not all primary caregivers provide PCC, educators should adhere to the dedicated referral
network developed within the strategy, because preconception care is only offered by a
minority of caregivers.14
• The training of the peer educators should be tailored to individual needs. It should include

194
 practical training and observation of a PCC consultation to increase educator’s familiarity and
appreciation of such services.
• Decisions should be made regarding the target group. Within this study municipalities targeted
potential social influencers of prospective mothers (e.g grandmothers).There is a rationale for
this approach however it may also take longer before such an approach sorts effectiveness
because target groups are reached indirectly.
• Future research should tailor preconception health messages to fit the needs of the potential
sub-groups amongst participants. Social marketing strategies could provide a framework for
this.21 Peer educators could include an individualized approach in their peer education sessions
- by applying a generic risk assessment tool.22-24 Identification of one or more risks might
motivate women to visit preconception consultations.
Supplementary figure 1 provides a renewed organizational model.

ETHICAL CONSIDERATIONS
The HP4ALL PCC sub-study has been approved by the Medical Ethical Committee of the Erasmus
Medical Center of Rotterdam (MEC 2012-425).

CONFLICTS OF INTERESTS
Danielle van Veen is associated to ‘the foundation of peer educators’ as an advisor. This foundation
is the foundation that effectuated the preconception health education in one of the 6 participating
municipalities. Sabine van Voorst, Vera Schölmerich, Carissah Stewarts, Eric Steegers and Semiha 9
Denktaş have no conflicts of interest to declare.

Implementation of peer health education

FUNDING
This work was supported by the Dutch Ministry of Health, Welfare and Sport [grant number 318804].

ACKNOWLEDGEMENTS
We would like to thank all peer educators, and all HP4All program managers for their pursuit to
realize the Peer Health intervention in their municipality and their readiness to participate in the
present evaluation.

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8. Temel S, van Voorst SF, de Jong-Potjer LC et al., The Dutch national summit on preconception care: a summary of defini-
tions, evidence and recommendations. J Community Genet. 2015;6:107-15. Epub 2014/11/15.
9. Hosli EJ, Elsinga J, Buitendijk SE et al., Women’s motives for not participating in preconception counseling: qualitative
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tion of preconception care in Leiden, the Netherlands] 2011 TNO/CH 2011.048.
11. van Voorst SF, Vos AA, de Jong-Potjer LC et al., Effectiveness of general preconception care accompanied by a recruitment
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Childbirth 2014;14. Epub 31 July 2014.
13. EURO-PERISTAT. PERISTAT II study. European perinatal health report. Better statistics for better health for pregnant women
and their babies in 2004. 2008.
14. van Voorst SF, Plasschaert SC, de Jong-Potjer LCA et al., Current practice of preconception care by primary caregivers in the
Netherlands. Eur J Contracept Reprod Health Care 2016;21(3):251-258.
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tation: a how-to guide. Health Promot Pract 2005;6:134-47. Epub 2005/04/28.
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SUPPLEMENTARY FIGURE 1: An improved organizational model for preconception health education.

Municipal policy maker

· formulation of policy within policy memorandum
· frame peer health education to meet existing health priorities
· allocation of budget

Field coordinator(s)
Managing peer educators
· Selection of educators
· Organization of training
and certification
· Monitoring educators
individual development
plan
Positioning peer educators Organizing peer
Evaluation
· Setting up networks health education
with PCC providing · Develop road · Number of women
midwives, GP’s and maps to recruit reached
hospitals target groups for · Characteristics of
peer health women reached
educations · Referral rates
· Monitoring data
collection

Peer Health educators

(Maintaining) expertise Network activities Organization of peer health
· Certification · Visitation of PCC education
· Individual development providers · Delivery of peer
plans education

Implementation of peer health education

197
 PART IV

4
BACK TO
POLICY & PRACTICE
GENERAL DISCUSSION
This thesis looks back upon the emergence of PCC in the Netherlands over the past decade – from
policy to practice and back. This chapter reflects upon principle findings and methodological
considerations. Recommendations are provided for translations of findings to policy and practice.

PART I – AGENDA SETTING AND EVIDENCE TO SELECT PCC AS

AN INTERVENTION
Aim of this part was to evaluate the policy process and to review the evidence which led to selecting
PCC as an intervention to reduce perinatal mortality. Key conclusions are summarized in Box 1.

Emergence of PCC as a standard component of perinatal care

Quantification of perinatal mortality and
morbidity in European context and in BOX 1: Key features in policy regarding PCC(Part I)
national context led to a debate about the
organization of perinatal health care in the • After the emergence of the concept of preconception

Netherlands. This perinatal mortality debate
led to a sense of urgency to innovate in
the organization of perinatal health care.
Various measures were formulated from the
preconception period to early childhood. The
key finding of our policy analysis in Chapter
care in the 1980’s, preconception care was taken up in
the Netherlands as a standard component of perinatal
health care policy as of 2009.
• The perinatal mortality debate promoted adoption of
preconception care.
• Preconception care is largely based on evidence for risk
factors and expert opinions.

2 was that the preconception period was • Implementation of preconception care requires a ‘leap
of faith’ from involved practitioners and policy makers,
acknowledged as a standard care component
whilst activities in the preconception field continue to
in perinatal healthcare policy.
emerge and are evaluated iteratively.
Before the perinatal mortality debate,
decisions regarding introduction of PCC
had been stalled by policy makers. PCC was
mostly initiated for single preconception health issues (e.g. genetic testing for autosomal recessive
disorders) or for health issues amongst specific populations. Reason to decline the implementation
of PCC was that the infrastructure for PCC was lacking. The Ministry of Health commissioned the
Health Council of the Netherlands and the Steering group to evaluate the effectiveness and
outreach of PCC before decisions regarding implementation of PCC were made.1,2 Where prior
agenda setting had failed to reach adoption of PCC, the perinatal mortality debate succeeded to.
This is striking given that the concept of PCC already took flight in the 1980’s.3,4 Thus, it took three
decades before introduction of PCC was agreed upon a national policy level.
Internationally, PCC has often been introduced in response to unfavorable perinatal mortality
and or morbidity rates. The primary aim of programs is often to reduce perinatal mortality and
morbidity.5-11 However, literature regarding the extent to which these preconception research
initiatives are taken up into national perinatal health policy of developed countries is scarce. To my
knowledge, the largest body of literature derives from the United States. Here, perinatal mortality

202
rates led to a ‘prenatal care crisis.’ Prenatal care had been promoted as the answer to infant mortality
in the beginning of the twentieth century. Yet, by the end of the twentieth century US prenatal
care was not doing what obstetricians had promised it would do. This led to the questioning of
the ability of prenatal care to combat adverse pregnancy outcomes. This fed the idea that there
were certain drivers for adverse pregnancy outcomes that were resistant to antenatal care. This led
to theorizing about innovative ways to improve birth outcomes.4 One of the consequences was
the launch of ‘the National Preconception Health and Health Care Initiative (PCHHC) of the Centers
of Disease Control and prevention (CDC).12-14 This initiative has achieved that PCC was integrated
into three subsequent editions of the ‘Healthy People’ blue prints, as of 1980. These documents
of the United States Department of Health and Human Services outline the ten year objective
for improving health in the United States.15-17 This approach illustrates a life course approach for
improving population based health. In the Netherlands, such a research frame and consequent
performance indicators for preconception health are lacking.

Implementation of PCC in the light of its evidence: a leap of faith?

Chapter 2 and 3 shows that evidence for PCC mostly relies on association studies showing the
association between (single) risk factors and adverse pregnancy outcomes. Available evidence for
interventions mostly focusses on single preconception interventions rather than comprehensive
intervention programs. Intervention studies mostly report self-reported behavioural change as the
outcome, rather than the effect on perinatal outcome.
Implementing PCC has been referred to by some as taking a ‘leap of faith’ in the light of the
current evidence for the effectiveness of interventions.4,18 However, there are three strong
arguments to implement PCC. Firstly, effectiveness has been established for single preconception
risk interventions.19 Secondly, PCC provides opportunity for reproductive choices (Dutch Definition
of PCC, Chapter 3) and secondary beneficial effects on parental health.20 Lastly, it is unethical to 10
withhold the emerging scientific knowledge about the importance of the periconception phase

General discussion
from prospective parents.
For the time being an authority based approach is acceptable. Having expert meetings, such as
the one described in Chapter 3, can generate ‘expert approved PCC program’ complementary to the
available evidence and guidelines. 21,22
The challenge at hand, is providing a comprehensive delivery approach, as evidence for such
approaches is lacking. Delivery approaches need to be implemented and evaluated in real time
settings to provide the population with care and to refine care based on the generated evidence for
different approaches. The Dutch national government has promoted research by allocating budget
for a dedicated research program for PCC (the Netherlands Organisation for Health Research and
Development (ZonMW)) as well as for the study that forms the basis of this thesis: the Healthy
Pregnancy 4 All - PCC sub-study.

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PART II - DESIGNING AN INTERVENTION STRATEGY
The aim of part II was to provide insights in how a programmatic PCC intervention strategy can be
developed in high risk municipalities. Key features are summarized in Box 2.
The Healthy Pregnancy 4 All (HP4All) - PCC program is in line with contents of policy (Chapter
2), the expert meeting (Chapter 3) and the
available evidence (Chapter 3 and 4). The PCC
BOX 2: K
 ey features in the design of intervention
program is consistent to policy formulated in
approaches (Part II)
response to perinatal mortality with respect
to the fact that it aims to activate the public
• The Healthy Pregnancy 4 All program was a vehicle to
move the national policy towards a strategy for munici-
health care at municipal level to participate in
pal implementation of preconception care. PCC. The PCC program is in line with the view
• The PCC sub-study is in accordance with views of the of the field that dedicated actors are needed
field. to recruit women for PCC, that PCC should
• Given the limitations in the evidence of PCC, the PCC
be delivered in a comprehensive approach
sub-study is evaluated iteratively.
and that available tools should be used
• The creation of a dedicated field for preconception
care requires ‘multilevel intervention’ and ‘knowledge
(ZwangerWijzer). Lastly, given limitations of
brokering.’ the evidence highlighted in Chapter 4, the
effectiveness of the program is evaluated
iteratively.
To elaborate on the design of the HP4All – PCC sub-study from a broader perspective, I would
like to emphasize its ‘multilevel intervention’ strategy and refer to the method of ‘knowledge
translation.’ These are two emerging concepts in the public health care field.21-23
Multilevel interventions: HP4All is designed as a multilevel intervention program. Multilevel
interventions can broadly be defined as intervention programs with targets to create change at
more than one of the following levels: policy level, community level, organizational level, intra- or
interpersonal levels.21 The HP4All program targets four of these levels (see Table 1). The multilevel
intervention strategy is a strength of the HP4All study because it provides vertical integration of
PCC interventions. This provides benefits for PCC. Firstly, it allows utilizing socio-environmental
resources such as the local health care professionals and local health care facilities. Local health
care professionals could promote outreach of the intervention, as they might be trusted more by
the target population in the community when it comes to discussing pregnancy contemplation.24
Utilizing existing services is also important because PCC requires integration with other services.
E.g. in case of smoking, a woman can be referred to a local smoking cessation service. Local health
care providers may provide such services themselves or are likely to be acquainted with those
services within the reach of their patients. This could enlarge the effectiveness of the PCC strategy,
not to mention the effectiveness of other existing health care services. Another advantage of
the multilevel strategy is that it allows identification of socio-environmental factors if evaluation
of context factors is performed. This is important to fine-tune programs or to adopt strategies
in different settings. Lastly, perhaps the most important advantage of multilevel intervention
strategies is that it overcomes the volatile nature of health promotion programs. The strategy

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equips the local setting with expertise and experience which are likely to outlive the duration of the
program. This is important for sustainability. Equipping local settings with expertise is an essential
part of the cycle towards science based policy.25
Knowledge translation: It is commonly acknowledged that there is a gap between ‘what is
known and what gets done in practice.26,27 This gap is attributed to the large volume of scientific
knowledge findings which is difficult for policy makers and health care professionals to keep up
with and may be too complex to understand.28 It has even been estimated that it takes 17 years to
turn 14% of funded research into benefits to patient care.29 Basic elements for science based practice
are knowledge generation, knowledge exchange and knowledge uptake.28 Knowledge brokering
is a knowledge exchange strategy in which intermediaries, or ‘brokers’ function as mediators
between researchers and intended users to help them understand each other’s languages and

TABLE 1: HP4All as a multilevel intervention with knowledge brokering at different levels

LEVEL WORKING DEFINITIONS HP4ALL TARGET KNOWLEDGE BROKERS

Policy level “Larger systems possessing the Municipal Aldermen and health HP4All
means to control several aspects of care departments were made
the lives and development of their aware about their perinatal health
constituent subsystems.”31 situation and the need for PCC.

Community “Collectives of people identified The programs recruitment strate- HP4All, municipal pro-
level by common values and mutual gy targeted the awareness for the gram managers
concern for the development and need, benefits and availability of
well-being of their group or geo- PCC services amongst women of
graphic area (villages neighbour- reproductive age within the com-
hoods).”31 munity.
10
Organizational “Systems with a formal multi-ech- The availability of PCC was created HP4All, municipal pro-

General discussion
level elon decision process operating in and the quality of PCC was ensured gram managers
pursuit of specific targets (schools, by means of training of health care
companies, professional organisa- professionals and the use of tools.
tions)” 31

Intrapersonal “Persons and small groups with Not applicable Not applicable
level whom the at-risk people associate”
(family friends).” 31
Interpersonal “Characteristics of the individual The recruitment approaches and HP4All, municipal pro-
level such as knowledge, attitudes, be- the preconception consultations gram manager, recruit-
haviour, self-concept, skills, etc.”32 targeted improved knowledge and ment partners, PCC pro-
behavioural change regarding the viders
uptake of PCC consultations and
behavioural change

Adapted from Scholmerich V.L.N. and Kawachi I, Multilevel interventions: theory and practice.33

205
eliminate barriers to the use of scientific knowledge.30 In the HP4All program knowledge brokering
was an ongoing mechanism to drive the actions of the project at the different levels (see Table 1).
Available evidence suggests that knowledge brokering promotes the use of research evidence to
inform decision-making while quality of the
knowledge used is improved.30
BOX 3: K
 ey features in the implementation of PCC
(Part III)
PART III – IMPLEMENTATION
OF PCC • Awareness about preconception health and PCC was
low amongst Aldermen, Municipal health services,
The aim of part III was to evaluate youth health services, GP’s and high amongst midwives.
implementation of PCC with its stakeholders. • Networks for programmatic PCC are underdeveloped.
It provides insights regarding awareness • The role of the Dutch public health system in PCC needs

for PCC, existing networks for PCC, the to be further explored.

• The private nature of the topic of conception is a barrier
promotion of PCC, the delivery of PCC and
for all stakeholders to approach women; yet women
context factors.
are more lenient than we think they are with regards to
what is acceptable in the promotion of PCC.
Awareness of PCC • Individual comprehensive PCC is only delivered at a
Low awareness of PCC was a recurring small scale and tends to be single risk factor focussed

theme throughout the studies of this thesis rather than comprehensive.

and during the introduction of the HP4All
preconception program in the field.
Firstly, the need for PCC was hardly
known amongst most Aldermen, Municipal
health care departments and GP’s. They were
• It is not necessary that all GP’s and midwives deliver
PCC, however it is necessary that they all recruit for PCC.
• GP’s need to utilize the identified moments in routine
care to recruit for PCC consultations.
• PCC needs to be delivered in a more uniform way; risk
assessment tools and ‘blue prints’ for risk factors can

surprised when they were confronted with provide a solution.

• Context factors: Lack of reimbursement for general PCC
the high perinatal mortality and morbidity
and fragmentation of care in the Dutch tiered health
rates as presented in Chapter 5. They
system impede implementation of PCC.
provided the explanation that they didn’t • Indicators need to be developed to monitor implemen-
have access to their loco-regional perinatal tation of PCC.
health statistics. Although prevalence’s of
perinatal morbidity and mortality was high at
municipal levels, experienced prevalence within the whole case load by GP’s and midwives was
said to be too low to be tangible. Most stakeholders said awareness of local perinatal mortality and
morbidity numbers provided the urgency to participate in HP4All.
Secondly, the field was unaware of PCC. Although Aldermen, Municipal health care departments,
Youth Health Care (YHC) professionals and GP’s were aware of some single preconception risk
factors (e.g. folic acid), many were unaware of delivery of PCC. On the other hand, midwives were
very aware of comprehensive PCC consultation services. This seems to be because when the
midwifery guideline for PCC was made available, training in delivery of PCC was provided and it
was integrated into the curriculum of midwives training programs. Other than publication in the

206
scientific journal of Dutch GP’s followed by one temporary e-learning tool there have been no other
structural efforts (e.g. training) to inform and train GP’s about PCC.34 In order to achieve delivery of
PCC, awareness of the need for PCC and skills to either deliver or refer for PCC are essential.
Awareness of women in the Netherlands regarding the need and the availability of PCC is low.
This has been experienced in prior Dutch programs and is a recurring finding in international studies
as well.35-38 This should be an important target point for campaigns and recruitment strategies.

Existing networks for programmatic PCC

Within the HP4All - PCC strategy collaboration was needed between public health care and curative
health care. These networks were generally underdeveloped. The peer health education component
(Chapter 9) illustrated this: women were not referred for PCC consultations to participating GP’s
and midwives providing PCC despite the program implementation strategy. Some women had
been referred beyond the network. However, it is not simply the referral action that counts; it is
about referring women to the right professionals that counts.
The commissioners of the HP4All study foresaw that it would be challenging to achieve
collaboration between public health care and curative health care. Such collaboration is a generic
requirement for the current perinatal health care field and for women’s’ health services. With regards
to PCC it is important that efforts to create networks between public and curative health care are
made and/or maintained. The different stakeholders have different expertise and resources and
can therefore have a synergistic effect in PCC initiatives. Lastly collaboration is important to embed
efforts in the cycle of policy making – intervention design – implementation and evaluation. This
increases effectiveness and sustainability.

Promotion of PCC
Manuscripts in this thesis and experiences in setting up the HP4All program provide insights in the 10
recruitment roles of stakeholders.

General discussion
Prior to the HP4All study, the public health system had a very minimal role in preconception
health promotion (Chapter 2, figure 1) and no role in recruiting women for PCC. Sending
invitational letters was not new for municipalities (e.g. youth vaccination program or cervical
cancer screening). Yet, when it came to an active role in inviting women for PCC there were more
barriers than foreseen. The main barriers to send the municipal letter was the fears of hurting
feelings of sub- or infertile women and to meddle with such a personal topic as conception as a
governmental institute. These concerns were taken seriously and the letter was adapted so 11 of
the 14 municipalities agreed to send the letter.
Youth Health Care (YHC) practitioners were asked to personally inform women of a PCC
consultation by midwives or GP’s during consultations for their children. Barriers to implement this
strategy was that it would be in contradiction to the trending working approach to deliver care
depending on individual risks (a high-risk approach) and demand (a demand driven approach). The
‘high-risk approach’ conflicts with the scientific finding that no less than 98% of the primary care
population has one or more risk factors for which a PCC consultation is indicated.39 Furthermore

207
the prevalence of preconception risk factors is likely to be higher in the designated area’s
(Chapter 3). The demand driven approach conflicts with the fact that the demand for PCC and
the preconception health literacy are both low (Chapter 7 and 8). Consumer research highlights
that the high-risk approach is not in line with the preferences of women, who found it acceptable
to be told about preconception care regardless of their risks (Chapter 8). In other words these
approaches are not according to the so called ‘every women every time’ approach that is necessary
for recruitment.40 The program failed in achieving active personal referral by YHC: YHC activities
were mainly restricted to providing an information leaflets and posters in waiting rooms.
Where the municipal letter and referral by YHC were not meant to selectively target women with
a pregnancy wish, peer health intervention was meant to. However, process evaluation showed
that only a minority of women that attended peer health education sessions had a pregnancy
wish (Chapter 9). Implementation evaluation explained that this was because preconception
health was introduced into existing meetings about other health topics, rather than that women
were specifically recruited. This was not the programs recruitment strategy. However, perhaps
reaching people influential to (future) mothers can indirectly sensitize women to the importance of
preconception health and other recruitment activities. Effectiveness of such a strategy would need
to be evaluated.
GP’s are accustomed to opportunistic approaches in pointing out preventive health care
issues.41 We discovered several moments (e.g. pregnancy wish, fertility matters, negative pregnancy
test, hereditary diseases and after miscarriage) during which both GP’s and women find suitable
to talk about PCC. These moments are currently underutilized by GP’s (Chapter 7 and Chapter
8). Furthermore, GP’s need to be aware of the importance of the availability and the benefit of a
comprehensive consultation besides only pointing out risk factors. In other words, they need to
abandon their ‘single risk factor approach’ and translate their activities to the explicit delivery of
PCC consultations.
It is not common practice for GP’s to send invitational letters for PCC (Chapter 7). GP’s insisted
they could exclude patients from the mailing because of fear to hurt patients’ feelings in certain
circumstances (again because of subfertility or adverse life-events). The program agreed that
exclusion criteria could be applied, if reasons for exclusion were accounted for. This meant we had
to deviate from the HP4All protocol as the program intended to send letters to all women aged 18
and up to 41 (Chapter 6). 21930 letters were sent in 32 of the 49 general practices. The letter is likely
to have missed some women with a pregnancy wish. The Parents to Be Study demonstrated, that
GP’s simply cannot predict who has a pregnancy wish.42
Prior to the program, midwives that delivered PCC have set up recruitment for their own
practice (e.g. with leaflets). Furthermore, the professional organization provides a list of midwives
that are certified to deliver PCC. Experience was that prior small scale attempts have only yielded
small scale uptake of PCC, if at all. This has been experienced as demotivating. Efforts at larger scale
are necessary and were welcomed.
Overall, the implementation of the HP4All recruitment interventions was a first step in
engaging the field in the recruitment for PCC. More important, it led to discussions about the roles

208
of the different stakeholders and which target population would be suitable for them to address.
A trunk line was the fear that the recruitment approach would be inappropriate. At the same time
the number of complaints from recipients was very limited. Chapter 8 also showed that women
find many approaches acceptable if explained adequately. This should encourage stakeholders to
participate in recruitment approaches for PCC in the future. The effectiveness of the strategy and
process evaluation needs to provide data to further adopt and refine strategies.

Delivery of PCC
Outside the scope of HP4ALL, the delivery of comprehensive preconception care by GP’s and
midwives only occurs at a very small scale (Chapter 7). This could be due to the underlying opinions
as to who is responsible for PCC. A substantial number of GP’s and midwives believe they are not
primarily responsible for delivering PCC (Chapter 7). I am convinced that if there is a guideline from
a professional organization, the specific topic belongs to its professionals. However, regardless of
responsibility, whether a GP or midwife delivers PCC should depend on whether they feel capable
to do so. HP4All showed us that it required training and conducting 5-10 PCC consultations to feel
capable of delivering PCC. Therefore, it might be a solution to concentrate the actual delivery of PCC
consultations to one or two GP’s or midwives in a community. This will require more collaboration
between professionals. Such collaboration is currently felt to be limited.43
Lack of uniformity of the contents of PCC (Chapter 7) is problematic. I believe uniformity starts
with standardized risk assessment. The utilization of risk assessment tools needs to be increased,
especially amongst GP’s.
To date, comprehensive PCC has not been implemented within the YHC setting in the
Netherlands. During the HP4All PCC study, some YHC professionals indicated they were willing
to deliver comprehensive PCC consultations themselves. To explore this, we adapted the
questionnaire in Chapter 7 to investigate Youth Health Care physicians’ activities and attitudes 10
towards PCC. A low response rate (8%; n=88/1088) brought us to exclude the results from this

General discussion
chapter. 22% of the YHC physicians found themselves primarily responsible for PCC. Although this
result should be interpreted with caution, it highlights that there may be some ambition to deliver
PCC. This was confirmed within a recent consensus meeting with YHC professionals, although it
also became apparent that YHC professionals foresee barriers in the delivery of PCC.44 As women
said that they prefer to receive PCC from a GP or midwife, preference of women to receive PCC
from YHC physicians would need to be evaluated (Chapter 8). The role of YHC is currently further
investigated in the sequence program of HP4All: “Healthy Pregnancy 4 All 2.”45
Preconception care is now an isolated care entity within perinatal health care. There is a need
to have continuity between preconception care and antenatal care. This is important to ensure
women’s compliance with the preconception health plan during pregnancy and for efficiency
(there is a large overlap in the risk assessment during pregnancy for instance). This requires that
a preconception health file is integrated into antenatal files and that communication between
professionals improves. Changing the organization of perinatal care was the main point of
departure after the societal perinatal mortality debate described in Chapter 2. One can also argue

209
that it could be integrated ‘vertically’ with other preventive health services. In the US the desire to
integrate PCC into ‘well women visits’, has been expressed.46 Long-term effectiveness of these visits
are unknown, however they are thought to promote delivery of preventive services.47 Revisiting the
preventive health scheme across a woman’s lifespan can make delivery of care more efficient.

Context factors
The Dutch tiered health care system provides challenges in the organization of perinatal health care
(Chapter 2). Preconception care is complex, requires extensive risk assessment and subsequent
intervention. In this process, it is likely to come across risk factors that exceed the competence of
the respective professional. Therefore, collaboration is necessary across the tiers and there is a need
for multidisciplinary care pathways to deliver preconception health interventions. To overcome
the challenges of the tiered system during pregnancy, HP4All’s other sub-study developed care
pathways for risks. Many of these care pathways could be adapted to the preconception phase and
hence promote uniformity of care.48
Before and during implementation of the HP4All study, PCC consultations were not reimbursed
within basic health insurance packages. Consequently, many providers did not deliver PCC or
transferred costs into bills for the patients. However, women are generally not willing to pay for
PCC (Chapter 8). During the HP4All program a temporary reimbursement plan had to be created to
achieve delivery of PCC without financial barriers. It is an excellent development that the 4 largest
health insurance companies now reimburse PCC.49
Up to 2010 the monitoring of perinatal health was mostly seen as a responsibility of the
curative field. Public health care partners often had no access to perinatal health statistics. If we
want the public health care system to become more involved in perinatal care, university medical
centers need to collaborate with municipalities in the evaluation of loco-regional perinatal health.
Indicators for the receipt and delivery of PCC need to be formulated and integrated within existing
data registries to monitor PCC.

Methodological considerations
There are some overall methodological considerations in this thesis.

Study populations
This thesis is based on data that reflects all stakeholders that have a role in PCC. This is one of the
strengths of this thesis, yet we have some considerations regarding the sampling of data from
these stakeholders.
The reflection upon the agenda setting of PCC (Chapter 2) mostly relies on the document
analysis within the database of the Dutch government, which included ‘PCC’ in its search. Key–
informants were selected if they were deemed to have had a significant role in the perinatal
mortality debate or if they were mentioned by other key-informants. By applying this selection
criterion, we could only identify key–informants for PCC agenda setting if they participated in the
agenda setting of PCC in relation to perinatal mortality. Therefore, we may have missed events

210
which had a specific role in the agenda setting for PCC. This limits the verification of findings
regarding the agenda setting of PCC.
A selection bias may be present with regards to sampling of practitioners. Professionals with
more affiliation with PCC are probably more inclined to participate in the expert meeting (Chapter
3), the inventory about PCC activities (Chapter 7) and in the HP4All PCC study (Chapter 9). This
could have resulted in overinflation of results leading to more positive findings. It is challenging
to include women preconceptionally in preconception studies. Women planning to conceive
(excluding subfertile women) do not present themselves within networks where it is opportune to
enroll women into studies. Thus, we had to broaden the eligibility criteria for participation in our
consumer research (Chapter 8). Women were eligible if they did not exclude wanting a pregnancy
in the future. This might make findings more negative than when opinions about PCC were
assessed purely amongst women with an actual pregnancy wish.

Review of evidence for PCC

It is a strength that the literature reviews in Chapter 2 and Chapter 3 include observational studies
as well as case-control studies. Only including randomized controlled trials (RCT’s) is incorrect with
regards to PCC. It is not ethical to recruit women in the preconception period and not provide any
form of preconception intervention because of the evidence of several single risk factors.

The high risk approach

In the design of the Healthy Pregnancy 4 All program there was a need to roll out interventions in
municipalities with highest perinatal mortality and morbidity rates, because effectiveness of the
program was believed to be the largest there. This required developing an intervention approach
that targets populations which are known to be the most difficult to reach. Retrospectively, one
can argue whether this is the most practical approach as hard-to-reach groups require special 10
strategies. As an intervention strategy in general is lacking, it would have also been feasible to

General discussion
develop a strategy for the general population and to consequently fine tune the strategy to target
risk areas. This fine-tuning would then require audience segmentation and developing materials
together with members of the target population. Practical reasons set aside, it may well be that
introducing PCC amongst the general population first may increase acceptability of PCC amongst
the hard-to-reach women.

Effectiveness of recruitment strategy and individual PCC

This thesis does not include results regarding the effectiveness of the recruitment strategy and
preconception consultations. This is because the program implementation had to be extended to
be able to achieve the calculated sample size. This was given in by the fact that time remaining for
implementation was less because it took longer than expected to set up the programs. Furthermore,
it took more time as the program was rolled out in 14 municipalities instead of 6 municipalities.
After extending the inclusion period, it was closed In December 2014 because the target population
was not thought to be attainable and there was a lack of funding to further extend the inclusion

211
period. A total of 587 women had made an appointment for a PCC consultation, of which 259 (44%)
women participated in the study. This is below the calculated study size of 839 which means the
study is underpowered. Lack of statistic power is likely to be due to short inclusion period (one
municipality had an inclusion period of 9 months), insufficient implementation of the peer health
education recruitment component, and the youth health care recruitment component and limited
effectiveness of the recruitment strategies.
It proved that the by far the most important hampering factor in the HP4All study was related
to bringing about change within the health care system itself. Process evaluation is necessary to get
grip on to the challenges of implementation in the real time setting and to understand program
outcomes.

Process evaluation
A limitation of our approach to process evaluation (Chapter 9) is that the process evaluation is
conducted by HP4All itself. Evaluation by an external partner which would include evaluation of
the role of HP4All itself, would increase validity. Internationally, there is a need for evaluation of
implementation strategies so initiatives can learn what works or doesn’t work in various settings.

RECOMMENDATIONS
After reflecting upon findings in this thesis, the following recommendations apply to future PCC:
• Implementation and research of PCC need to occur within a continuous cycle of agenda
setting, intervention selection (or refinement), implementation and back translation to policy
and practice. This requires:
·· Keeping PCC on the policy agenda by providing numbers regarding PCC performance
indicators (e.g. % of pregnancies after PCC consultations, number or PCC consultations in
relation to women of reproductive age). Data collection could be integrated into the Dutch
Perinatal Registry and into the 4 annual municipal health inquiries which municipalities use
to identify health and socioenvironmental issues that need to be addressed in preventive
health care policy.
·· Iterative evaluation of implementation and effectiveness of PCC intervention strategies
·· Central coordination of PCC research initiatives according to predefined targets and expert
meetings to renew or refine intervention strategies.
·· Knowledge brokers need to be coordinated centrally so they can translate experiences in
implementation of PCC in different settings to new area’s in which there is no programmatic
approach to PCC.
• GP’s and midwives need to upscale and improve their PCC activities by:
·· utilizing moments in every day practice to point out availability of PCC consultations
·· integrating recruitment for PCC with other preventive health services
·· referring for PCC or deliver PCC themselves. If they deliver PCC themselves they need to
deliver PCC in a structured uniform way using established risk assessment tools and

212
 blueprints for preconception risk factors agreed on by multidisciplinary working groups.
• The role of the public health care system needs to be further defined. Trends to deliver
preventive health care in the Netherlands according to a ‘demand driven’ or ‘high risk’ approach
is not applicable to recruitment for PCC.
• To increase effectiveness of programmatic PCC all stakeholders need to invest in mutual
collaboration.
• Recruitment models for PCC need to include a multiple hit approach to ensure women are
reached out to at several moments in their reproductive lives.
• PCC strategies need to be tailored locally. This can be done by involving the respective target
group prior to implementation and by adapting best practices from other settings.

CONCLUSION
This thesis provides reflection upon the agenda setting for PCC, selection of interventions for
PCC, the design of a programmatic approach for PCC and implementation of PCC by different
stakeholders in PCC. Despite evidence about PCC risk factors, comprehensive PCC only occurs at
a small scale. By implementation of programmatic PCC within the HP4All study, PCC has been
implemented at a large scale as a multilevel strategy. Main challenge of delivering programmatic
PCC is to achieve the required organizational changes needed amongst all stakeholders within
communities. Awareness needs to increase, mind shifts are required, knowledge and expertise
needs to increase and new collaborations need to develop. It appears that these prerequisites need
to be further developed before a PCC program for the general population can be effective.

REFERENCES
10
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General discussion
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General discussion
48. Vos AA, van Voorst SF, Waelput AJ, et al. Effectiveness of score card-based antenatal risk selection, care pathways, and
multidisciplinary consultation in the Healthy Pregnancy 4 All study (HP4ALL): study protocol for a cluster randomized con-
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detail/vergoedingen-voor-iud-implantatiestaafje-en-preconceptiezorg/2032.

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SUMMARY / SAMENVATTING
ENGLISH SUMMARY
Current perinatal care is inadequately addressing risk factors in early pregnancy. By the time
antenatal care commences (at best in the 8th week of gestation) the foetus has already been
exposed to risk factors during crucial events in its development. These risk exposures are associated
to perinatal mortality and morbidity. Over the past decade most focus has been set on perinatal
mortality as indicator of perinatal health care quality. However, mortality is only the tip of the
iceberg: being born with perinatal morbidity can give rise to disease and illness in childhood
and adulthood. Preconception care (PCC) addresses risk factors before conception and therefore
reduces chances of perinatal morbidity and mortality. It is the most primary form of prevention
there is. Perinatal care should include PCC in order to promote perinatal health, which is the first
step in having a health society.
Countries are facing challenges regarding how PCC can be delivered effectively with sufficient
outreach. In the Netherlands, it has been advocated to deliver PCC in the form of individual
consultations to the general public.
This thesis looks back upon events in the past decade and research regarding the organization
of individual PCC within the Dutch primary care and public health system. This thesis is presented
according to the following cycle in which health policy is ideally developed. Firstly, a health issue
reaches the agenda and interventions are selected (Part I); an intervention approach is designed
(Part II); lessons are drawn from implementation and evaluation in practice (Part III) and findings are
back translated to policy and practice (Part IV).
Hence the title of this thesis: ‘Preconception care - from policy to practice and back’.

Part I – Agenda setting and intervention selection

Preconception care was selected as an intervention during the policy process described in Chapter
2. Intervention selection is ideally performed in light of available evidence and with experts in the
field (Chapter 3 and 4).
Chapter 2 provides a chronologic reconstruction of how the relatively unfavorable perinatal
mortality and morbidity rates of the Netherlands compared to other European countries (EURO-
PERISTAT reports) and the inequalities in perinatal health (Chapter 5) led to policy reform between
2004 and 2010. The perinatal health problem became a national health issue with a high priority.
This was promoted by the quantification of perinatal mortality and morbidity, the nature of the
topic and involvement of the media and the political field. Where the typical Dutch organization
of perinatal care was undisputed prior to the debate, the debate led to the recognition that how
perinatal care was organized needed to be changed. The debate engaged a broad range of actors
beyond the curative field. Proposed measures reflected the multidisciplinary collaboration of
actors. The importance of preventive approaches in which socioeconomic determinants of perinatal
health was acknowledged and this emphasized the important role of municipalities. The scope of
prenatal care was (re)defined as care including the preconception period until early childhood.
Involvement of municipalities was deemed important to address socioeconomic determinants
of perinatal health. A contextual factor impeding the policy process was the tiered organization

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of perinatal health care in the Netherlands. The different tiers have fundamentally different
perspectives regarding the ideal organization of perinatal health care. The Healthy Pregnancy 4 All
(HP4All) study is one of the products of this policy process. It is in accordance with the several key
aspects of the newly formulated perinatal health policy identified in Chapter 2.
Aim of the Dutch national summit on PCC (2012) was to contribute to policy making and
implementation by evaluating current evidence, gaps in knowledge and opinions of experts in the
field. Chapter 3 presents the results of this meeting. Highlights concern:
• Definition: The definition of PCC was broadened with regards to three aspects. Firstly, PCC is a
program or a set of interventions rather than a single intervention. Secondly, non-medical risks
are addressed besides medical risks. Thirdly, the definition now highlights that counselling and
informed decision making are important goals of PCC.
• Organizational approaches: There was consensus to categorize PCC into collective measures,
general individual PCC and specialist individual PCC. The role of different health care
professionals within general and specialized preconception care needs to be defined.
• Target groups: It was acknowledged that a broader range of actors needed to become involved
in advocating PCC to target groups.
• Contents: PCC was expanded to address more risk factors. Future research should point out how
caregivers should prioritize in interventions when multiple risk factors are present.
• Risk assessment instruments: There was consensus to adopt a generic risk assessment instrument.
The field stated the need to optimize and validate existing tools or to develop new tools.
Chapter 4 shows there is evidence for the effectiveness of preconception interventions addressing
nutrition, alcohol, smoking and folic acid intake. Generalizability of these findings may be limited
since interventions were mostly delivered as single measures and amongst selected populations.
Evidence is deemed sufficient to provide lifestyle recommendations to the general public while
effectiveness of interventions is evaluated iteratively.

Part II - Development of an intervention strategy

11
This section provides the design of the Healthy Pregnancy 4 All (HP4All) study.

Summary / Samenvatting
Chapter 5 shows how we decided upon the areas for dissemination of the intervention study.
We contemplated that municipalities with highest perinatal mortality and morbidity would benefit
most from intervention. We performed a geographic analysis of perinatal mortality and morbidity
after which 14 municipalities were selected. All approached municipalities agreed to participate in
the HP4All study. The HP4All study was designed to consist of interventions that aimed to reduce
risks in early pregnancy: a programmatic approach to preconception care and early standardized
antenatal risk assessment during early pregnancy. The Preconception care sub-study and the Risk
Assessment sub-study were set up to evaluate the effectiveness of both interventions.
Chapter 6 describes the rationale and design of the HP4ALL Preconception care sub-study. The
intervention consisted of an individual PCC consultation delivered by general practitioners (GP’s)
and midwives in the community. The PCC consultation consisted of an initial consultation during
which risk assessment was performed and a tailored management plan was made. A follow-up

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consultation took place 3 months later. Additionally, a recruitment strategy was designed to target
women to utilize the PCC services. Invitational letters were sent by municipal public health services
and participating general practices and women were referred by Youth health care centers and
peer health educators.
The effectiveness of the PCC sub-study is evaluated with a cohort study. Primary outcome
regarding the effectiveness of consultations is behavioural change regarding preconception
health behaviours (folic acid supplementation, cessation of smoking, alcohol consumption and/
or drug use). Primary outcome regarding the effectiveness of the recruitment strategy is outreach
expressed in the number of women recruited and characteristics of women approached. Sample
size calculation resulted in a study size of 839 women. Participants were enrolled between February
2013 and December 2014.

Part III - Evaluation of Implementation

This part reflects on what can be learned from different stakeholders in preconception care:
midwives and general practitioners (Chapter 7), women contemplating pregnancy (Chapter 8) and
municipal public health partners including peer health educators (Chapters 9).
Chapter 7 provides the results of a cross-sectional survey to evaluate current activities,
perceptions and prerequisites of GP’s and midwives regarding individual PCC consultations.
• Current activities: This study confirms that systematic delivery of PCC consultations only
occurs at a small scale. Activities GP’s and midwives are mostly opportunistic: risk factors are
pointed out and questions about preconception risks are answered upon confrontation in daily
practice. Explicit promotion and delivery of an actual individual PCC consultation only occurs
occasionally. The content of the delivered PCC is not uniform amongst caregivers.
• Perceptions: Respondents’ perceptions about PCC consultations are generally positive. Midwives
see themselves as the professionals who are primarily responsible for the delivery of PCC. GP’s
are ambivalent as to whether GP’s or midwives are primarily responsible for PCC. Respondents
are willing to increase their promotion of a comprehensive PCC consultation during various
moments in routine care. Providers are willing to use risk assessment tools, which promote
uniformity of consultations.
• Prerequisites: Respondents find more training about PCC, staff support, promotion of PCC and
adequate reimbursement to be prerequisites for future delivery of PCC.
Chapter 8 presents findings of consumer research. This is the first step towards social marketed
preconception care consultations. The philosophy of social marketing is that products are designed,
so both the needs of women and the system are met. A qualitative study design was used to assess
preferences across the four essential components (or the four P’s) of the social marketing: ‘Product,
Place, Price and Promotion’. The most striking finding was the low knowledge of the purpose and
benefits of the product (PCC). Perceived needs of women depend on their obstetric history and
concerns about fertility and parenthood. PCC is not addressing these concerns. Low knowledge of
the product emphasizes the importance of promotion. Women express that they want to make the
informed decisions whether they utilize PCC services. They see the primary care setting as the most

220
suitable place to be informed about PCC. We identify several specific occasions for professionals to
bring up PCC. However, women also prefer to be informed about PCC, regardless of their risk factors
because they might have risk factors they don’t know about themselves. This should support
health care professionals to become more active in the promotion of systematic PCC consultations.
Regarding place characteristics, women find the community based primary care system as the
most suitable care echelon. Lack of reimbursement is a dilemma for women that see the benefits of
PCC yet have financial means to attend PCC.
Chapter 9 provides an in-depth process evaluation of the peer health education strategy in
HP4All. In HP4All we integrated peer education from the public health care domain into a strategy
to promote uptake of PCC consultations. Strategy was that peer educators would refer participants
of peer education sessions to preconception consultations centers in the community. This strategy
was adopted in 7 municipalities.
The implementation criteria were met in 4 of the 7 municipalities. However, implementation
at overall program level was insufficient because outreach was insufficient. Peer educators did
reach underserved women. However the seldomly referred women. Qualitative data provided that
this was due to lack of time to set up the program as intended, lack of adherence to the target
population and lack of effective working relationships between peer educators / municipal health
services and the centers delivering PCC consultations. The strategy needs to be refined to draw
conclusions regarding the effectiveness of the strategy.

Part IV - Translation of findings back to policy and practice

In Chapter 10 we reflect upon the main findings of this thesis, as well as the strength and limitations
of the study. We conclude by providing recommendations regarding the organization of general
PCC within the Dutch health care system.

11

Summary / Samenvatting

221
NEDERLANDSE SAMENVATTING
De huidige perinatale zorg schiet tekort in het tijdig aanpakken van risicofactoren in de vroege fase
van de zwangerschap. Tegen de tijd dat antenatale zorg start (in het meest gunstige geval rond
de achtste week van de zwangerschap) heeft de foetus al tijdens kritische ontwikkelingsprocessen
blootgestaan aan risicofactoren die geassocieerd zijn met perinatale mortaliteit en morbiditeit.
Bijvoorbeeld roken of het gebruik van geneesmiddelen door de moeder. Lange tijd was er focus
op perinatale mortaliteit als indicator voor de kwaliteit van perinatale zorg. Perinatale mortaliteit is
echter slechts het ‘topje van de ijsberg.’ Geboren worden met perinatale morbiditeit is geassocieerd
met een grotere kans op het ontwikkelen van ziekten in het latere leven. Omdat men met
preconceptiezorg risicofactoren aanpakt vóór de conceptie, kunnen deze risicofactoren aangepakt
worden als zij nog geen (of minimale) schade hebben kunnen berokkenen. Preconceptiezorg is
daarom de meest primaire vorm van preventie die er is. Prenatale zorg dient de preconceptiefase
te benutten voor gezondheidsbevordering voor moeder en kind. Dit ligt ten grondslag aan een
gezonde maatschappij.
Verschillende landen staan voor de uitdaging hoe preconceptiezorg effectief en met voldoende
bereik geleverd kan worden. In Nederland hebben we het adagium om algemene preconceptiezorg
in de vorm van individuele consulten in de eerste lijn aan te bieden aan de algemene populatie.
Dit proefschrift blikt terug op de gebeurtenissen van het afgelopen decennium en op
onderzoek met betrekking tot de organisatie van individuele preconceptiezorg in de eerste lijn
en in het publieke gezondheidsbestel. Het proefschrift is ingedeeld volgens de chronologische
stappen van gezondheidsbeleid. Eerst komt een gezondheidsprobleem op de agenda, worden
er interventies geselecteerd (Deel I). Vervolgens wordt er een interventiebeleid ontwikkeld
(Deel II), wordt implementatie van de interventie geëvalueerd (Deel III) en vervolgens worden
bevindingen terugvertaald naar beleid en praktijk (Deel IV). Vandaar de titel van dit proefschrift:
‘Preconceptiezorg – van beleid naar praktijk en terug.’

Deel I – Agendasetting en interventie selectie

Preconceptiezorg is geselecteerd als interventie tijdens het beleidsproces dat beschreven wordt
in Hoofdstuk 2. Interventie selectie wordt idealiter uitgevoerd in het licht van beschikbaar bewijs
voor de interventie en met experts uit het veld (Hoofdstuk 3 en 4).
De beleidsanalyse in Hoofdstuk 2 geeft weer hoe de relatief ongunstige cijfers voor perinatale
mortaliteit en morbiditeit hebben geleid tot een vernieuwd perinataal gezondheidsbeleid.
Perinatale gezondheid kreeg hoge prioriteit toen bleek dat Nederland ongunstig scoorde op
perinatale sterftecijfers ten opzichte van omliggende landen. Daarnaast werd in Nederland een grote
ongelijkheid gezien in perinatale gezondheid. De kwantificering van het gezondheidsprobleem, de
aard van het onderwerp en de betrokkenheid van de media en de politiek hebben bijgedragen aan
de prioriteitstelling om maatregelen te nemen. Er ontstond bewustzijn dat er verandering nodig
was in de manier waarop perinatale zorg georganiseerd werd. Maatregelen werden geformuleerd
voor de hele perinatale zorgketen, die gedefinieerd werd als zorg vanaf de preconceptie fase
tot zorg in de vroege kinderjaren. Voorgestelde maatregelen reflecteerden de inbreng van de

222
verschillende actoren, afkomstig van verschillende disciplines. Zo kwam de nadruk te liggen op
preventieve maatregelen en het rekening houden met socio-economische determinanten van
perinatale gezondheid. Hierbij werd een rol toegekend aan gemeenten. Het ‘lijnen systeem’ van de
Nederlandse zorg en de verschillende perspectieven van verloskundigen en gynaecologen die met
elkaar in strijd waren bemoeilijkten het beleidsproces.
De ‘Healthy Pregnancy 4 All’ studie is één van de initiatieven die zijn oorsprong vindt in het
beschreven beleidsproces. Het programma komt op meerdere punten tegemoet aan speerpunten
in het beleid dat tijdens deze periode geformuleerd is.
Hoofdstuk 3 beschrijft de resultaten van de Nederlandse expertmeeting over preconceptiezorg
in 2012. Deze expertmeeting heeft geresulteerd in de volgende kernpunten:
• De definitie: De definitie van preconceptiezorg is verbreed. Het is gedefinieerd als een
programma of een set van maatregelen in plaats van een interventie t.a.v. een enkele risicofactor.
Verder omvat de definitie nu ook dat preconceptiezorg niet-medische risicofactoren adresseert
en dat counseling en geïnformeerde besluitvorming doelstellingen zijn.
• Organisatorische benaderingen: Er was consensus om organisatorische benaderingen voor
preconceptiezorg onder te verdelen in collectieve maatregelen, algemene individuele
preconceptiezorg en specialistische individuele preconceptiezorg. Er dient afgestemd
te worden wat de rol is van verschillende zorgverleners in algemene en specialistische
preconceptiezorg.
• Doelgroepen: Er moeten meer disciplines betrokken raken bij het promoten van
preconceptiezorg onder doelgroepen.
• Inhoud: De inhoud van preconceptiezorg is uitgebreid. Onderzoek moet uitwijzen hoe er
geprioriteerd moet worden in interventies als er meerdere risicofactoren aanwezig zijn.
• Risicoselectie instrumenten: Er is consensus om één generiek risico instrument te gebruiken.
Beschikbare tools moeten geoptimaliseerd en gevalideerd worden of er moeten nieuwe
instrumenten ontwikkeld worden.
Hoofdstuk 4 toont de effectiviteit voor interventies in de preconceptiefase gericht op voeding,
11
alcohol, roken en foliumzuurinname. Generaliseerbaarheid van deze bevindingen naar de

Summary / Samenvatting
algemene populatie kan beperkt zijn omdat de interventies geïmplementeerd zijn als losse
interventies (in plaats van in een programma) en omdat veel studies binnen geselecteerde
populaties zijn uitgevoerd. Toch is de bewijsvoering voldoende om de betreffende leefstijl-
interventies te implementeren, bij voorkeur iteratief.

Deel II - Ontwikkeling van een interventie strategie

Dit deel geeft inzicht in de ontwikkeling van de ‘Healthy Pregnancy 4 All (HP4All) studie.
Hoofdstuk 5 laat zien hoe we de gebieden voor de uitrol van het HP4All programma hebben
geselecteerd. Er werd verondersteld dat de gemeenten met de hoogste perinatale mortaliteit en
morbiditeit de meeste winst zouden behalen met de interventies. Na een geografische analyse
van perinatale mortaliteit en morbiditeit werden 14 gemeenten geselecteerd. Alle benaderde
gemeenten hebben ingestemd met deelname aan het HP4All programma. In lijn met het bewijs dat

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embryonale gezondheid cruciaal is voor verloop en de uitkomst van de zwangerschap, omvat de
HP4All studie twee interventies: 1. een programmatische aanpak voor individuele preconceptiezorg
en 2. systematische risicoselectie in de zwangerschap. Effectiviteit van de interventies werd binnen
tegelijk lopende zorgexperimenten geëvalueerd.
Hoofdstuk 6 gaat in op de opzet en de onderbouwing van het HP4All preconceptiezorg
experiment. De interventie bestond uit een individueel preconceptiezorgconsult bij deelnemende
huisartsen en verloskundigen. Het consult bestond uit een eerste bezoek waarin risicoscreening
plaatsvond en een plan werd gemaakt om bestaande risicofactoren te bestrijden. Drie maanden
later vond er een vervolgconsult plaats. Om het gebruik van de preconceptiezorgspreekuren te
bevorderen werd een wervingsstrategie toegepast. Er werden uitnodigingsbrieven door gemeente
en door deelnemende huisartsen verstuurd. Consultatiebureaus, Centra voor Jeugd en Gezin
(CJG’s) en speciaal opgeleide voorlichters preconceptiezorg verwezen vrouwen naar de spreekuren.
De effectiviteit van het programma wordt geëvalueerd door middel van een cohort studie. De
primaire uitkomstmaat om de effectiviteit van de consulten te meten is gedragsverandering t.a.v.
enkele preconceptionele leefstijl factoren (foliumzuur suppletie, stoppen met roken, alcohol en
drugsgebruik). De effectiviteit van de wervingsstrategie wordt gemeten aan de hand van het bereik
van het programma. Er werd berekend dat er 839 vrouwen nodig zouden zijn om de effectiviteit
van preconceptiezorg te kunnen evalueren. Inclusie vond plaats tussen februari 2013 en december
2014.

Deel III - Evaluatie van implementatie

In dit deel wordt nagegaan wat er geleerd kan worden van de verschillende ‘stakeholders’ in de
algemene preconceptiezorg. Hoofdstuk 7 geeft inzicht over implementatie door verloskundigen
en huisartsen, Hoofdstuk 8 geeft inzicht in de visie van vrouwen over het preconceptiezorg consult
en Hoofdstuk 9 over de rol van de publieke gezondheidszorg en gezondheid voorlichters.
Hoofdstuk 7 geeft inzicht in de huidige activiteiten, percepties en randvoorwaarden van
huisartsen en verloskundigen m.b.t. preconceptiezorgconsulten.
• Actiteiten: Dit onderzoek bevestigd dat preconceptiezorgconsulten slechts op kleine
schaal aangeboden en uitgevoerd worden. Activiteiten van huisartsen en verloskundigen
zijn voornamelijk ad hoc: ze wijzen vrouwen te vaak alleen op risicofactoren als ze ermee
geconfronteerd worden tijdens de alledaagse zorg. Expliciet aanbod van een apart
preconceptiezorgconsult gebeurt nagenoeg niet. Wanneer het wel plaatsvindt, blijkt de inhoud
niet uniform.
• Percepties: Zorgverleners waren positief over preconceptiezorgconsulten. Verloskundigen
zien zichzelf als de primair verantwoordelijke zorgverlener voor preconceptiezorg, terwijl
huisartsen ambivalent zijn of ze zelf primair verantwoordelijk zijn of dat verloskundigen dit zijn.
Respondenten waren bereid om gebruik te maken van diverse momenten in alledaagse zorg
om vrouwen te wijzen op de mogelijkheid van een preconceptiezorgconsult. Verder waren zij
bereid om gebruik te maken van instrumenten voor risicoscreening, hetgeen de inhoudelijke
uniformiteit van consulten zou bevorderen.

224
• Randvoorwaarden: (Meer) training over preconceptiezorg, personele ondersteuning, strategieën
om vrouwen naar preconceptiezorgconsulten toe te leiden en adequate vergoeding zijn
randvoorwaarden voor het aanbieden van preconceptiezorg in de toekomst.
Tot slot wordt in Hoofdstuk 8 het resultaat van het consumentenonderzoek gepresenteerd. Dit
is de eerste stap richting preconceptiezorg volgens het ‘social marketing’ principe. De filosofie
van deze strategie is dat een zorgproduct zo wordt ontworpen dat het tegemoet komt aan de
doelen van zowel de doelgroep als het zorgsysteem. D.m.v. kwalitatief onderzoek zijn preferenties
binnen de 4 domeinen (of de 4 P’s) van social marketing onderzocht: ‘Product, Plaats, Prijs en
Promotie.’ De meest opvallende bevinding t.a.v. het product was de lage kennis over het doel
en de potentiele baten van het preconceptiezorgconsult. Of vrouwen noodzaak ervaarden voor
een preconceptiezorg consult bleek vooral af te hangen van de verloskundige voorgeschiedenis
van vrouwen en van hun vragen over vruchtbaarheid en ouderschap. De laatste elementen
komen niet standaard aan de orde binnen preconceptiezorg consulten. Beperkte kennis over het
product benadrukt het belang van promotie strategieën. Vrouwen willen graag geïnformeerde
keuzes maken over het wel of niet gebruik maken van het preconceptiezorg consult. Ze zien de
huisartsen en verloskundigen setting als de meest geschikte plaats om geïnformeerd te worden
over een preconceptiezorg consult. Vrouwen vinden diverse aanleidingen in dagelijkse zorg van
hun huisarts of verloskundige een goed moment om gewezen te worden op het preconceptiezorg
consult. Bovendien vinden vrouwen het ook acceptabel om geïnformeerd te worden als ze geen
evident risicoprofiel hebben, dit kan immers pas blijken na uitgebreide risicoscreening. Dit zou
zorgverleners moeten ondersteunen om een actievere houding aan te nemen in het promoten van
preconceptiezorg consulten. Vrouwen hebben een voorkeur voor preconceptiezorg in de eerste lijn,
bij huisartsen en verloskundigen of in gezondheidscentra. Moeten betalen voor preconceptiezorg
zadelt vrouwen met beperkte financiële middelen op met een dilemma t.a.v. bezoeken van het
preconceptiezorgconsult.
Hoofdstuk 9 bevat de procesevaluatie van de voorlichting preconceptiezorg binnen HP4All.
Waar het gebruikelijk is om een voorlichtingsstrategie te implementeren binnen de context van
11
de publieke gezondheid, is met de HP4All strategie getracht om een brug te slaan naar zorg

Summary / Samenvatting
binnen de curatieve setting: de huisartsen en verloskundigen die preconceptiezorg aanbieden in
het project. Doel van voorlichters in de HP4All strategie was dat speciaal opgeleide voorlichters
deelnemers van groepsvoorlichtingen zouden verwijzen naar preconceptiezorgconsulten.
Hiermee vervullen voorlichters een brugfunctie naar individuele eerstelijns preconceptiezorg.
De strategie is geadopteerd in 7 gemeenten. Vier van de 7 gemeenten voldeden aan de
implementatiecriteria. Op programma niveau was de implementatie onvoldoende omdat het
bereik onvoldoende was. De moeilijk te bereiken groep werd bereikt, maar er werd niet verwezen
naar preconceptiezorgconsulten. Kwalitatief onderzoek gaf inzicht in verklaringen. Voornaamste
redenen waren tijdsgebrek, het niet vasthouden aan de afgesproken doelgroep en het gebrek
aan effectieve samenwerking relaties tussen voorlichters, gemeenten en de praktijken die
preconceptiezorg aanboden in het programma. De strategie moet verbeterd worden om conclusies
te trekken over de effectiviteit van voorlichters als verwijzers naar individuele preconceptiezorg.

225
Deel IV - Terug naar beleid en praktijk
Tot slot reflecteren we in Hoofdstuk 10 over de hoofdbevindingen en de methodologische sterkte-
en zwaktepunten van dit proefschrift. We vertalen bevindingen terug naar aanbevelingen voor de
organisatie van algemene preconceptiezorg in het Nederlandse zorgsysteem.

226
11

Summary / Samenvatting

227
ADDENDUM

LIST OF ABBREVIATIONS
AUTHORS AND AFFILIATIONS
LIST OF PUBLICATIONS
PHD PORTFOLIO
ABOUT THE AUTHOR
DANKWOORD
LIST OF ABBREVIATIONS
ABS Absolute rate
CCKL Coördinatie Commissie ter bevordering van de Kwaliteit beheersing
CDC Centers for Disease Control and Prevention (CDC)
CDT% Carbohydrate deficient transferrin
CI’s confidence intervals
cm centimeter
CPZ Commission for Perinatal Health (CPZ)
EtG ethylgluconeride
GBA Gemeentelijke Basis Administratie (GBA) or the municipal administrative records
GP General Practitioner
HP4All Healthy pregnancy 4 All
INEQ Inequality rate
IQR Interquartile range
kg Kilogram
PeTH phosphatidylethanol
PCC Preconception care;
PRN Perinatal Registry Netherlands;
R4U Rotterdam reproductive risk reduction score card;
SD Standard deviation
SES Socio-economic status;
SGA Small for gestational age;
STND Standardized rate
YHC Youth Health Care

230
AUTHORS AND AFFILIATIONS
From the Erasmus University Medical Centre, Department of Obstetrics and Gynecology, Division of
Obstetrics and Perinatal Medicine, Rotterdam, the Netherlands:
Gouke J. Bonsel, Chantal A. ten Kate, Semiha Denktaş, Lieke L.C.A. de Jong-Potjer, Sophie C.N.
Plasschaert, Jashvant Poeran, Eric A.P. Steegers, Carissah J.C. Stewarts, Sevilay Temel, Danielle W. van
Veen, Sabine F. van Voorst, Amber A. Vos, Adja A.J.M. Waelput

From the Erasmus University College, Department of Social Sciences, Rotterdam, the Netherlands:
Semiha Denktaş, Vera L.N. Schölmerich

From the Institute of Health Policy and Management, Erasmus University, Rotterdam, the Netherlands:
Erwin Birnie

From the VU University Medical Centre, EMGO Institute for Health and Care Research, Department of
Clinical Genetics, Section of Community Genetics, Amsterdam, the Netherlands:
Martina C. Cornel

From the Albert Schweitzer Hospital, Department of Obstetrics and Gynaecology, Dordrecht, the
Netherlands:
Sabina R. Rombouts - de Weerd

From the Boston University School of Medicine, Department of Family Medicine, Boston Massachusetts,
United States of America:
Brian W. Jack

A
ADDENDUM

231
LIST OF PUBLICATIONS

Manuscripts in this thesis

S.F. van Voorst‡, A.A. Vos‡, E.A.P. Steegers, S. Denktaş. Analysis of policy towards improvement of
perinatal mortality in the Netherlands (2004–2011). Social Science and Medicine. 2016 May;157:156-
64.
‡ shared first authorship

S.Temel, S.F. van Voorst, L.C. Potjer, A.J. Waelput, M.C. Cornel, S. de Weerd, S. Denktaş, E.A.P.
Steegers. The Dutch national summit on preconception care: a summary of definitions, evidence
and recommendations. Journal of Community Genetics. 2015 Jan;6(1):107-15.

S. Temel, S.F. van Voorst, B.W. Jack, S. Denktaş, E.A.P. Steegers. Evidence-Based Preconceptional
Lifestyle Interventions. Epidemiologic Reviews. 2014;36:19-30. doi: 10.1093/epirev/mxt00

S. Denktaş, J. Poeran, L.C. de Jong-Potjer, S.F. van Voorst, A.A. Vos, A.J.M. Waelput, E. Birnie, G.J.
Bonsel, E.A.P. Steegers. The Healthy Pregnancy 4 All Study: Design and cohort profile. BMC
pregnancy and Childbirth 2014.14:253.

S.F. van Voorst, A.A. Vos, L.C. de Jong-Potjer, A.J.M. Waelput, E.A.P. Steegers, S. Denktaş. Effectiveness
of general preconception care accompanied by a recruitment approach: protocol of a community-
based cohort study (the Healthy Pregnancy 4 All study). BMJ Open. 2015; 5.

S.F. van Voorst, S.C.N. Plasschaert, L.C. de Jong-Potjer, E.A.P. Steegers, S. Denktaş. Current practices
of Preconception care by Primary caregivers in the Netherlands. European Journal of Contraception
and Reproductive Health Care. 2016 Jun;21(3):251-8.

S.F. van Voorst, C.A. ten Kate, L.C. de Jong – Potjer, E.A.P. Steegers1, S. Denktaş. Developing social
marketed individual preconception care consultations: which consumer preferences should it
meet? Health Expectations. 2017 Jan; 00:1–8.

S.F. van Voorst, V.L.N. Schölmerich, C.J.C. Stewarts, D.W. van Veen, E.A.P. Steegers, S. Denktaş.
Implementation of community-based peer health education strategy for preconception care.
Manuscript submitted for publication.

232
Publications not included in this thesis:

Four Barriers in the Delivery and Uptake of Preconception Care: A Qualitative Study
H. Ismaili M’Hamdi, S.F. van Voorst, W. Pinxten, I.D. de Beaufort, M. Hilhorst, E.A.P. Steegers. Maternal
and Child Health Journal. 2017 Jan;21(1):21-28.

S. Denktaş, A.J.M. Waelput, S.F. van Voorst, A.A. Vos, G.J. Bonsel, L.C. de Jong Potjer, E.A.P. Steegers.
Healthy Pregnancy 4 All, Tijdschrift voor gezondheidswetenschappen. 2012, 90 (8), pp 479-483.

A.A. Vos, S.F. van Voorst, A.J.M. Waelput, L.C. de Jong, G.J. Bonsel, E.A.P. Steegers, S. Denktaş.
Effectiveness of score card-based antenatal risk selection, care pathways, and multidisciplinary
consultation in the Healthy Pregnancy 4 All study (HP4ALL): study protocol for a cluster randomized
controlled trial. Trials. 2015, 16:8.

S.F. van Voorst, S. Temel, I.A. Aalhuizen, S. Denktaş, E.A.P. Steegers. Handboek vrouwspecifieke
geneeskunde, Hoofdstuk 18 Preconceptiezorg [Guidebook female specific healthcare: Chapter 18
Preconception care], Prelum Uitgevers, red B.C.J.M. Fauser e.a., 2013.

S. Denktaş, L.C. de Jong-Potjer, A.J.M. Waelput, S. Temel, S.F. van Voorst, A.A. Vos, V.L.N. Schölmerich,
G.J. Bonsel, J. van der Kooy, C. Quispel, V.J.J. Poeran, I.A. Peters, M. Vuijkovic, R. Bakker, E.A.P. Steegers.
Preconception care a review of the literature, Report written for the World Health Organization
meeting on Preconception care, Geneva. 2012.

S.F. van Voorst, N.P. Johnson. How to improve your IVF succes rates, ‘Chapter 11 How to manage
hydrosalpinges,’ Cambridge University Press, 2011.

N.P. Johnson, S.F. van Voorst, M.C. Sowter, A. Strandell, B.W.J. Mol. Surgical treatment for tubal
disease in women due to undergo in vitro fertilisation. Cochrane Database of Systematic Reviews
2010, Issue 2.
A
N.P. Johnson, S.F. van Voorst, M.C. Sowter, A. Strandell, B.W.J. Mol. Tubal surgery before IVF. Human
ADDENDUM

Reproduction update. 2011, 17 (1): 3.

T.E. Nieboer, N.P. Johnson, S.F. van Voorst, B.W.J. Mol, K.B. Kluivers. Surgical approach to
hysterectomy for benign gynaecological disease. Cochrane Database of Systematic Reviews 2009,
Issue 3.

233
Manuscripts in preparation

A.A. Vos, S.F. van Voorst, A.G. Posthumus, A.J.M. Waelput, S.Denktaş, E.A.P. Steegers. Implementation
of scorecard-based antenatal risk selection, care pathways, and interdisciplinary consultation: the
Healthy Pregnancy 4 All Risk Assessment study. Manuscript submitted for publication.

M.E. Hopman, M. Essink – Bot, A.N. Rosman, S.F. van Voorst, M.P. Fransen. Recruitment for
preconception counseling: A scoping study among women with low health literacy. Manuscript
submitted for publication.

S.F. van Voorst, M.K. Sijpkens, A.A. Vos, L.C. de Jong- Potjer, S. Denktaş, E.A.P. Steegers. Evidence
of the adoption and implementation of a preconception care program in the Netherlands (the
Healthy Pregnancy 4 All study). Manuscript in preparation.

M.K. Sijpkens, S.F. van Voorst, L.C. de Jong- Potjer, S. Denktaş, A.R. Rosman, L.C.M. Bertens, A.P.
Verhoeff, E.A.P. Steegers. Effectiveness of preconception care recruitment strategies (the Healthy
Pregnancy 4 All study). Manuscript in preparation.

234
A
ADDENDUM

235
PHD PORTFOLIO

PhD candidate: Sabine Francisca van Voorst

Institution: Erasmus MC
Department: Obstetrics and Prenatal Medicine
PhD Period: September 2012 to March 2015
Promotors: Prof.dr. E.A.P. Steegers, Prof.dr. S. Denktaş
Copromotor: dr. L.C. de Jong - Potjer

1. PhD training Year ECTS

Courses
• Introduction to Biostatistics for Clinicians (NIHES). 2012 0.7
• Regression analysis for Clinicians (NIHES). 2012 1.4
• SPSS course for research in care (Erasmus MC). 2012 0.5
• Scale Development (Karma Short Course). 2012 0.5
• Basiscursus Regelgeving en Organisatie voor Klinisch 2013 1.0
onderzoekers (BROK; Erasmus MC).
• Principles of analysis in Epidemiology (NIHES). 2014 0.7

Conferences
Presentations at International conferences

• 2nd European Conference on Preconception Care and Health, 2012

Rotterdam, the Netherlands:
·· Oral presentation: “A systematic Review on 1.0
Preconceptional Risk factors: New Risk Factors to Address
in Preconception Care.”
·· Flash presentation: “Consensus on a definition on 0.5
Preconception Care in the Netherlands.”
·· Flash presentation: “Experimenting with Programmatic 0.5
Preconception Care in 14 High Risk Municipalities in the
Netherlands.”
·· Flash Presentation: “A Systematic Review on 0.5
Preconceptional Risk factors: New Risk Factors to Address
in Preconception Care.”

236
• Satellite meeting, Developmental Origins of Health and 2012 0.5
Disease (DOHAD), Rotterdam, the Netherlands:
·· Poster presentation: “Experimenting with Programmatic
Preconception Care in 14 High Risk Municipalities in the
Netherlands.”
• 61st Annual meeting of the Society for Gynaecologic 2014 0.5
investigation, Florence, Italy:
·· Poster presentation: “General Preconception Care in
Primary and Youth Health Care – Implementation in the
Netherlands.”
• Ministerial Conference of Women’s Health, Rome, Italy: 2014 1.0
·· Invited speaker: “Preconception Care: A Life course
approach.”
• 3rd European Conference on Preconception Care and Health, 2016 1.0
Uppsala, Sweden:
·· Oral presentation: “Consumers preferences as a point
of departure for social marketed preconception care
consultations.”

Presentations at national conferences, seminars or research

meetings
• HP4All local kick-off meetings. 2012 1.0
• HP4All progress meetings with the field. 2013 - 2014 1.0
• HP4All program manager meetings. 2012 - 2014 1.0
• Research meetings of the Department of Obstetrics and 2012 - 2013 1.5
Prenatal Medicine: three presentations on the design and
progress of the HP4All Preconception care sub-study.
• HP4All Symposium Preliminary findings of the Preconception 2014 1.0 A
care sub-study.
ADDENDUM

Attended meetings/ Conferences

• Attending weekly research meetings of the Department of 2011 - 2014 5.0
Obstetrics and Prenatal Medicine and giving 3 presentations.
• Annual RCOG Wladimiroff Symposia. 2011 - 2014 0.5
• Ready for a Baby (Klaar voor een Kind) End Symposium. 2013 0.3
• Symposium Urban Perinatal Health (SCEN). 2011 0.3

237
PHD PORTFOLIO (CONTINUED)

2. Teaching Year ECTS

Lecturing
• Guest lecture NIHES course: Urban Perinatal Health and 2012 1.0
Health Care. Lecture “Measuring Health Care Utilization –
amongst minorities.”
• Guest lecture NIHES course: Maternal and Child Health. 2012 1.0
Lecture “Preconception Care.”
• Guest Lecture Erasmus Faculty of Medicine: Minor: circle of 2012 1.0
life. Lecture “Preconception Care.”
• Guest Lecture Erasmus University College (EUC): Introduction 2014 1.0
to the social sciences. Lecture: “Application of sociology in
Health Care: Andersen’s model for utilization of Health care
services.”
• Annual Guest Lecture at the VAR (Rotterdam School of 2012 - 2014 3.0
Midwifery). Lecture: “Preconception Care.”

Trainings
• Practical at HP4All’s participating General Practices and 2012 - 2013 1.0
Midwife practices. Practical: implementing tools for
Preconception Care (ZwangerWijzer, PreconceptieWijzer).
• Practicals within the Peer Health Educators training. 2013 1.0
• Round table discussion about Preconception Care at the 2015 1.0
`Wereld café.´ VAR (Rotterdam School of Midwifery).
• Practical about Preconception Care at the Erasmus Faculty of 2015 0.5
Medicine: Minor: Mystery of creation.

Supervision activities
• Supervising elective research programs: S. Plasschaert, C. 2012 - 2014 4.0
Stewart.
• Supervising research: C. ten Kate. 2013 - 2014 1.0

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Other
• Obtaining approval by the Medical Ethics committee for 2012 1.0
the WMO associated multicenter study (Protocol title:
Project Healthy Pregnancy 4 All, Aanpak babysterfte in
Nederlandse gemeenten: ontwikkeling en implementatie van
zorgexperimenten. Deel A: Zorgexperiment Preconceptiezorg
MEC- 2012-425).
• Obtaining approval by the Medical Ethics committee for 2013 1.0
the not-WMO associated study (“Women’s preference for
individual Preconception Care” (MEC-2013-586)
• Answering questions of the City council about Municipal 2014 0.5
invitations for preconception care
• Obtaining approval to receive data from the Municipal 2012 - 2014 0.5
Administrative records of all 14 HP4All municipalities.
• Peer reviewing an article for a scientific journal 2014 0.5
• Collaboration with PREGEVA study (Dr. M. Franssen) 2013-2014 0.5

A
ADDENDUM

239
240
ABOUT THE AUTHOR
Sabine Francisca van Voorst was born as the first of a twin pregnancy in Rotterdam, the Netherlands
(1984). As the daughter of a Civil Engineer and a French teacher, she spent the first years of her
upbringing abroad in Burkina Faso, Indonesia, Curaçao and Suriname. At the age of 12 she
repatriated to The Hague, where she graduated from the Aloysius College in 2002.
From 2002 – 2008 she went to Medical School at the University of Maastricht. In her final year,
she combined a clinical internship with a research elective at the department of reproductive
health of the Maxima Medical Center in Veldhoven and at the Cochrane Menstrual disorders and
Subfertility group in Auckland, New Zealand. She graduated as a Medical Doctor in 2008.
After her study Sabine worked as a junior resident at the department of Obstetrics and
Gynaecology of the Reinier de Graaf Gasthuis in Delft and in the Erasmus Medical Center in
Rotterdam. Between 2011 and 2015 Sabine was a PhD Candidate within the Healthy Pregnancy 4 All
study. In accordance to the hypothesis that the course of pregnancy and its outcome is influenced
by events before and in early pregnancy, this study consisted of two pillars: preconception care and
systematic risk assessment with medical and non-medical care-pathways. Sabine was dedicated to
the design, implementation and evaluation of the preconception care sub-study. This study was
the point of departure for her thesis.
In November 2015 Sabine started her residency in Obstetrics and Gynaecology.
Sabine is married to Ronald Koekkoek, they live in The Hague.

A
ADDENDUM

241
DANKWOORD
Over een paar dagen gaat mijn proefschrift dan echt naar de drukker. Een moment om terug te
kijken op de afgelopen jaren.
Het is waar dat je vooraf niet kan bedenken wat je promotie je allemaal gaat brengen. Tijdens
Healthy Pregnancy 4 All (HP4All) heb ik de de kans gekregen om de hele verloskundige zorgketen
en de samenwerking met de publieke gezondheidszorg te verkennen. Die samenwerking met veel
verschillende mensen in deze keten heeft geleid tot het resultaat dat voor jullie ligt. Zonder een
groot aantal mensen was dit nooit zo goed gelukt en was het zeker niet zo leuk geweest.

Beste Prof.dr. Steegers, beste Eric, mijn eerste promotor. Allereerst bedankt voor het vertrouwen
om mij als promovenda aan te stellen. Ik ben blij dat ik met HP4All een stukje heb mogen bijdragen
aan jouw grotere puzzel van de sociale verloskunde en preconceptiezorg. Ik heb het gewaardeerd
dat er ruimte was voor verwondering over de belevenissen in het veld. Verder leverde het veldwerk
regelmatig onverwachte scenario’s op die niet zelden methodologische gevolgen hadden. Waar ik
hobbels zag, zag jij kansen en oplossingen. Tijdens de laatste fase van mijn promotie heb je mij de
ruimte gegeven om mijn promotie op mijn manier af te maken. Hartelijk dank voor je begeleiding.

Prof.dr. Denktaş, Semiha abla; “het is klaar!” Jouw programmaleiding en je dagelijkse begeleiding
was onmisbaar. Ik bewonder hoe je de projectgroep op koers hield en ons het grote plaatje voor
ogen bleef houden. Je hebt me geleerd vast te houden aan mijn doelen maar flexibel te zijn in
mijn methoden. Zo heb je mij ook gestimuleerd om mijzelf verschillende onderzoekstechnieken
eigen te maken. Je hebt een prettige balans tussen heel hard werken en heel hard lachen. Dat
jij hoogleraar zou worden was slechts een kwestie van tijd. Je oratie was voor mij een feest van
herkenning van wat ik de afgelopen jaren van je heb mogen leren. Ik ben er trots op om een van je
eerste ‘promovendi batch’ te zijn en hoop je nog regelmatig te blijven zien.

Dr. L.C. de Jong – Potjer, beste Lieke, beste copromotor, als promovendus in preconceptiezorg trad
ik in jouw voetsporen. Dit project was jou dan ook op het lijf geschreven. Je hebt een uniek talent
om mensen te enthousiasmeren. Dat was cruciaal voor de huisartsen participatie in het project en
net zo belangrijk om mij op sommige momenten op koers te houden. Het feit dat je op zoveel
fronten actief bent en je verwondert over van alles en nog wat gaf leuke stof voor de gesprekken
onderweg. Groningen was toch eigenlijk zo ver nog niet! Het project vergde meer dan de vier
dagdelen waarop je niet als huisarts werkte. Ik ben je dankbaar voor je flexibiliteit en toewijding,
waarmee we er altijd uit kwamen. Je bent een fijn persoon, ik ben dankbaar voor je wijze raad op
verschillende momenten en bovenal hoop ik nog regelmatig gezellig met je bij te kletsen!

Geachte leden van de promotiecommissie, ik wil u allen bedanken voor uw tijd en inzet voor de
beoordeling van dit proefschrift.

242
Zonder ‘veld’ geen veldwerk. Ik wil graag alle deelnemende verloskundigen, huisartsen, GGD’en,
CJG’s en voorlichters preconceptiezorg bedanken voor het deelnemen aan HP4All. Het viel niet
altijd mee om iets heel nieuws of de HP4All manier te gaan doen, naast de bestaande drukte in de
praktijk. Dank dat ik een kijkje mocht nemen in jullie alledaagse praktijk en dat we konden sparren
over de implementatie van preconceptiezorg. In het bijzonder dank ik onze lokale projectleiders:
Renee Stoffels (Groningen), Marieke Bunnik (Enschede), Gerdine Fransen en Mieke van der
Biezen (Nijmegen), Bea Zeelen (Heerlen), Miek van der Ham (Tilburg), Shanty Badal (Schiedam),
Evy Hochheimer (Utrecht), Arnoud Verhoeff, Brigitte Heusschen (Almere), Stefan Wiggers
(Almere) en Annette de Graaf (Den Haag). Bedankt voor het ‘gidsen’ en de leuke bezoekjes aan
jullie gemeenten!

Uiteraard dank ik al mijn co-auteurs voor hun bijdragen aan de artikelen in dit proefschrift. In
het bijzonder mijn 3 studenten: Chantal ten Kate, Carissah Stewart en Sophie Plasschaert. Ik
kreeg energie van onze samenwerking en ben er trots op dat onze samenwerking tot jullie eerste
artikelen heeft geleid!

Beste HP4All-1 collega’s: Amber Vos, Adja Waelput, Danielle van Veen en Ageeth Rosman. We
refereerden vaak aan onze HP4All tijd als een toneelstuk waarvan het script ‘kwijt’ was. Zonder alle
scenes op te halen kan ik zeggen dat we er zeker wat moois van hebben geregisseerd!
Beste Amber, ik had mij geen betere evenknie kunnen wensen. We zijn heel verschillend maar
vulden elkaar juist goed aan. Onze policy paper is daar het ultieme voorbeeld van. Ik bewonder je
focus en je doorzettingsvermogen. Jij promoveerde dan ook als eerste, wat ik mee mocht maken
als trotse paranimf. We blijven voorlopig nog even in hetzelfde schuitje zitten, als AIOS. Gezellig om
je nog bij cursussen te treffen en gewoon daarbuiten (ik kom gauw een beschuitje eten).
Beste Adja, dank dat je altijd beschikbaar was voor alle verloskundigen-georiënteerde vragen,
je literatuur adviezen, je notulen, je lach en je gezelligheid. Na afronding van HP4All-1 was je altijd
beschikbaar voor mijn vragen op afstand en moedigde je mij tot het laatst toe aan.
Beste Daan, je zorgde altijd voor leven in de brouwerij op de Westzeedijk! Dank voor je
toewijding aan de voorlichting Preconceptiezorg en je creativiteit om overal iets moois van te
maken! Het was fijn samenwerken met je!
A
Beste Ageeth, dank voor je ondersteuning bij het opzetten van de laatste gemeenten en de
inclusies. Dat gaf echt lucht om me op het schrijven te kunnen richten.
ADDENDUM

Beste Vera, ik noem je hier graag want je bent onze aangewaaide HP4All’er. Je was onze
vreemde eend, of zal ik zeggen zwarte kip. Daarmee gaf je onze groep diversiteit en (nog meer)
gespreksstof. Dat we goed konden samenwerken bleek uit onze VPG paper, dank voor je kritische
blik en input.

Beste Healthy Pregnancy 4 All -2’ers: Meertien Sijpkens, Jacky Lagendijk, Minke van de Minden,
Hiske Ernst, Ageeth Rosman en Adja Waelput. Fijn dat jullie de sociale verloskunde verder uitrollen
in andere delen van de verloskundige zorgketen!

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 Beste Meertien, mijn bloed, zweet en urine (samples ;-)) zijn letterlijk en figuurlijk die van
jou geworden. Ik had mij geen betere opvolger kunnen bedenken. Dank voor je trekkracht en je
detective werk, zoals je het zelf noemt. Dit allemaal naast je eigen HP4All-II werkzaamheden.
Beste Jacky en Minke bedankt voor alle gezelligheid als ik weer eens kom klussen met Meertien,
Succes met de afronding van jullie promoties!

Beste Westzeedijkers: beste Anke Posthumus, Babs van der Kooy, Chantal Quispel, Erwin Birnie,
Gouke Bonsel, Ingrid Peters, Jashvant Poeran, Kirsten Heetkamp, Marijana Vujkovic, Marisja
Scheerhagen, Mieke van Veen, Nynke de Groot en Sevilay Temel. Dank voor de fijne tijd met
gezellige lunches en koffiemomenten. Specifiek nog dank aan Babs, voor de hulp bij het opstarten
van steden en het knutselen met lab buisjes! Sevilay, leuk dat jij mijn voorganger was voor
preconceptiezorg en dat ik van jouw ervaringen heb mogen leren voor de ‘landelijke’ aanpak. En ik
kan het niet laten nog even te zeggen: wat hebben we lekker gereviewed!!
Beste Hafez je bent lekker kritisch en bekijkt het nut van preconceptiezorg vanuit nieuwe
perspectieven. Een eer dat ik met je mee mocht schrijven aan je 4 barriers manuscript. Succes met
de afronding van je promotie!
Beste (oud) collega-onderzoekers van de afdeling Verloskunde en Gynaecologie: Averil, Bas,
Babette, Caroline, Emilie, Evelyne, Irene, Kim, Leonie, Mathijs, Marit, Nicole, Nienke, Nina, Paulien,
Ruben, Wendy, Yvonne, Zoë, en ik ben ongetwijfeld iemand vergeten. Bedankt voor de gezellige
tijd, de woensdagochtenden, de ski-reis, de SGI en Uppsala!

Ankie! We Bedankt voor je vrolijkheid, hulpvaardigheid en je gezelligheid de afgelopen jaren! Ik

ben blij dat we dat als clustergenoten vast kunnen houden en gezellig handsfree kunnen beppen
over gynaecoloog worden of w.v.t.t.k!

Jolanda Claessens, hartelijk dank voor je ondersteuning bij het regelen van de promotoren-
overleggen en de zorg voor de logistiek rondom de afronding van dit proefschrift. Beste Brenda,
jouw vertrek was een verlies voor onze groep omdat je een enorme steun was bij de start van het
project. Ik ben echter heel blij voor je dat je zo goed op je plek voelt bij EUC.

Beste collega’s van het Ikazia, hartelijk dank voor de fijne ontvangst in Rotterdam-zuid!

Met dit boek wordt voor velen concreet en tastbaar wat ik de afgelopen jaren heb gedaan. Het was
echter nooit zo mooi geworden zonder Wouter! Dropbox verraadde vaak dat je er tot laat aan had
gewerkt. Je stond erop alle figuren mooi(er) te maken en tot het laatst was geen enkele moeite je te
veel. Ontzettend bedankt!

Lieve Jill, geheel conform onze stijl zijn we weer eens een mijlpaal vergeten: 15 jaar vriendschap!
Misschien weer eens tijd voor een weekendje weg naar de Caribbean?

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Lieve Noor, fijn dat je ook boven de rivieren bent komen wonen! Weliswaar niet meer in dezelfde
straat, maar gelukkig dichtbij!

Lieve oom Frans en tante Irene, het jaar wordt gemarkeerd met jullie strandbrunch en het
boerenkool diner. Ik ben blij dat jullie, net als bij andere grote momenten in mijn leven, erbij zijn
op deze dag!

Lieve Koekkoeks, ik prijs me gelukkig met jullie als schoonfamilie. Linda, Erwin en Ronald, zo
verschillend als jullie zijn, zo goed kunnen jullie elkaar aanvullen. Ik geniet ervan dat te zien en
op verschillende momenten in de afgelopen jaren heeft die kracht zich bewezen. Lin, bedankt dat
ik altijd bij je kan aankloppen voor afleiding, relativering of gewoon voor cafeïne. Tegelijkertijd
moet ik dan ook Tjan bedanken, dat je mij bijstaat als de Koekkoeks qua humor zichzelf weer weten
te overtreffen. Een blik is dan genoeg, daar moet ik dan op zich wèl weer om lachen. Juliette en
Victoria, ik geniet ervan om jullie “Bien” te zijn! Lieve Oma, bezoekjes aan Flakkee zijn altijd een
feest, ik hoop dat we dat nog lang mogen doen met jou, in goede gezondheid.

Lieve Romy! Onze gelijke start en vergelijkbare ‘route’ is een gezellige vriendschap geworden
waarin we veel met elkaar gedeeld hebben: dokteren, onderzoeken, trouwen en promoveren. Je
kan dan ook niet ontbreken op deze dag, als mijn paranimf. Ik hoop dat we nog veel leuke dingen
meemaken!

Liefste Veer, dank voor de vanzelfsprekendheid. Ik hoef bij jou nooit uit te leggen wat ik denk of
voel. Sterker nog, ik kan niet uitleggen hoeveel me dat waard is. Naast mijn zus ben je ook een
bevlogen huisarts. Die combinatie maakt dat ik er trots op ben dat je mij flankeert als paranimf!
Beste Daan, dank voor je altijd frisse kijk op zaken en voor de gezelligheid als ik bij jullie binnenval
of blijf plakken. En na september ben je officieel mijn zwager!

Lieve Pap en Mam, dit proefschrift benadrukt dat een goed begin het halve werk is en dat het
uitmaakt waar je wieg staat. Ik had me geen beter begin kunnen wensen (naast foliumzuur, heel
goed mam). Jullie moedigen mij altijd aan om vast te houden aan mijn doelen en ondersteunen
A
mij waar mogelijk. Een strategie die ik niet heb onderzocht in dit proefschrift, maar die jullie wel
geïmplementeerd hebben is het frequent verhuizen van de wieg. Het wonen in verschillende
ADDENDUM

landen met de verschillende mensen om ons heen heeft mij verrijkt. Dank voor de fijne thuisbasis,
gewoon om de hoek, waar ik altijd op terug kan vallen.

Liefste Ronald, met jou kan ik schaken op meerdere borden tegelijk. Verbouwen, promoveren, in
opleiding, grote reizen maken samen, trouwen .. het is allemaal gebeurd de afgelopen jaren. Toch
zijn de kleine dingen en de gewone dagen samen nog altijd het fijnst. Ik hou van je met heel mijn
hart!

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